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Phelan AK, Infante S, Barni S, Nurmatov U, Boyle RJ, Vazquez-Ortiz M. The Role of IgE Sensitization in Acute FPIES: A Systematic Review and Meta-Analysis. THE JOURNAL OF ALLERGY AND CLINICAL IMMUNOLOGY. IN PRACTICE 2025; 13:861-884.e3. [PMID: 39855467 DOI: 10.1016/j.jaip.2025.01.016] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 05/29/2024] [Revised: 01/13/2025] [Accepted: 01/14/2025] [Indexed: 01/27/2025]
Abstract
BACKGROUND Evidence on the role of immunoglobulin E (IgE) sensitization in acute food protein-induced enterocolitis syndrome (atypical FPIES) is limited. Initial reports claimed association with persistent disease; however, recent studies have not replicated this. OBJECTIVE To systematically review the relationship between sensitization to the culprit food(s) in acute FPIES and the outcome of follow-up oral food challenges. To assess the rates of sensitization, seroconversion (ie, switch from negative tests to sensitization), and phenotype switch to IgE-mediated food allergy over time in individuals with acute FPIES. METHODS Systematic review searching 10 databases. Studies of children and adults with an acute FPIES diagnosis assessing IgE sensitization to a culprit food at onset or follow-up measured by skin prick or serological test were included. RESULTS Of 1,830 studies identified, 53 were eligible including 3,514 participants. Ten studies had an analytical design assessing whether sensitization was associated with disease persistence, with 4 showing an association and 6 showing no association. In individuals with acute FPIES, the sensitization rate was 9.8% (95% confidence interval [95% CI 7.4%-12.1%; 34 studies, 2,587 participants, I2 = 82%); the frequency of seroconversion was 1.1% (95% CI 0.1%-2.1%; 9 studies, 673 participants, I2 = 32%); and phenotype switch occurred in 1.1% (95% CI 0.4%-1.7%; 14 studies, 935 participants, I2 = 0%) and 13% (95% CI 5.5%-20.5%, 12 studies, 93 participants; I2 = 18%) of sensitized participants. CONCLUSIONS We did not find consistent evidence for the relationship between IgE sensitization and FPIES persistence. We found phenotype switch to IgE-mediated food allergy is uncommon in acute FPIES. An IgE sensitization in FPIES does not have a clear relationship with clinical outcomes.
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Affiliation(s)
- Aisling K Phelan
- Paediatric Dietitians, St. Mary's Hospital, Imperial College London, London, UK.
| | - Sonsoles Infante
- Paediatric Allergy Unit, Hospital General Universitario Gregorio Marañón, Madrid, Spain
| | - Simona Barni
- Allergy Unit, Meyer Children's Hospital IRCCS (Scientific Institute for Research, Hospitilisation and Healthcare), Florence, Italy
| | - Ulugbek Nurmatov
- Division of Population Medicine, School of Medicine, Cardiff University, Cardiff, UK
| | - Robert J Boyle
- Section of Inflammation, Repair and Development, National Heart and Lung Institute. Imperial College London, London, UK
| | - Marta Vazquez-Ortiz
- Section of Inflammation, Repair and Development, National Heart and Lung Institute. Imperial College London, London, UK
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Ibrahim T, Argiz L, Infante S, Arasi S, Nurmatov U, Vazquez-Ortiz M. Oral Food Challenge Protocols in Food Protein-Induced Enterocolitis Syndrome: A Systematic Review. THE JOURNAL OF ALLERGY AND CLINICAL IMMUNOLOGY. IN PRACTICE 2025; 13:814-832. [PMID: 39746512 DOI: 10.1016/j.jaip.2024.12.033] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 04/03/2024] [Revised: 12/20/2024] [Accepted: 12/24/2024] [Indexed: 01/04/2025]
Abstract
BACKGROUND Oral food challenges (OFCs) are essential for the diagnosis and follow-up of acute food protein-induced enterocolitis syndrome (FPIES) because no diagnostic or prognostic biomarkers are available. However, the optimal OFC procedure remains unclear. OBJECTIVE This systematic review aimed to assess OFC procedures' design and clinical outcomes in patients with FPIES. METHODS We searched 10 databases for studies published in English between 1978 and February 2024 involving children or adults undergoing OFC for FPIES. Critical appraisal followed Effective Public Health Practice Project parameters. RESULTS In total, 52 studies met inclusion criteria, all observational studies. Of these, 35 were judged to have strong methodological quality. There was great heterogeneity in OFC procedures, particularly in cumulative dose, number, size, and timing between doses. Oral food challenge outcome reporting was often inadequate, especially regarding reaction symptoms and severity grading. In single-dose OFC protocols, most children reacted after at least 2 hours. Four small studies showed that a single dose of 25% of an age-appropriate portion was sufficient to trigger reactions in 80% to 100% of cases, and this was associated with less severe reactions. Owing to methodological heterogeneity and insufficient outcome reporting, further assessment of the OFC protocol characteristics associated with safer outcomes was not possible. CONCLUSIONS There is significant heterogeneity in FPIES OFC practices. Current recommendations for OFC procedures and outcome assessments have limitations and should be revisited, because this may affect patient safety and diagnostic accuracy. Future studies should focus on standardizing clinical outcomes and generating evidence to support safer, more accurate OFC protocols in FPIES.
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Affiliation(s)
- Tayseer Ibrahim
- Allergy and Immunology Division, Department of Medicine, Hamad Medical Corporation, Doha, Qatar.
| | - Laura Argiz
- Department of Allergy, Clínica Universidad de Navarra, Pamplona, Spain; RICORS Red De Enfermedades Inflamatorias (REI)- RD21/0002/0028, Madrid, Spain
| | - Sonsoles Infante
- Pediatric Allergy Unit, Hospital General Universitario Gregorio Marañón, Gregorio Marañón Health Research Institute, IiSGM, Madrid, Spain
| | - Stefania Arasi
- Allergy Unit, Department of Pediatric Medicine, Bambino Gesù Children's Research Hospital, IRCCS, Rome, Italy
| | - Ulugbek Nurmatov
- Division of Population Medicine, School of Medicine, Cardiff University, Cardiff, United Kingdom
| | - Marta Vazquez-Ortiz
- Section of Inflammation, Repair and Development, National Heart and Lung Institute, Imperial College London, London, United Kingdom
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Perriere A, Garcette K, Kalach N, Tounian P, Lemoine A. Food protein induced enterocolitis syndrome: French practices assessment in children. Arch Pediatr 2025; 32:114-119. [PMID: 39875216 DOI: 10.1016/j.arcped.2024.12.003] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/19/2024] [Revised: 09/23/2024] [Accepted: 12/08/2024] [Indexed: 01/30/2025]
Abstract
BACKGROUND Food protein-induced enterocolitis syndrome (FPIES) is a specific non IgE-mediated food allergy. The international consensus guidelines defined diagnosis criteria and management plan in 2017. OBJECTIVES To assess practices regarding FPIES in France and in French-speaking countries, according to those guidelines. METHODS AND SETTING We carried out a 22-question online survey to assess practices of specialised physicians (paediatricians, allergists, gastroenterologists, members of 2 French-speaking learning societies and/or working at hospitals in paediatric allergy units) following patients with FPIES between August 2019 and February 2022. RESULTS We received 92 replies to our survey, mostly from hospital practitioners following less than 10 patients with FPIES. Oral rehydration solution and/or Ondansetron were largely prescribed in the emergency kit (61/72, 84.7 % and 47/72, 65.3 % respectively). 20 practitioners declared never prescribing an emergency kit. There was some confusion when distinguishing between FPIES and an IgE-mediated food allergy, as suggested by the unnecessary prescription of an antihistamine (18/72, 25.0 %) and/or epinephrine (11/72, 15.3 %) in the emergency kit. An explanatory FPIES emergency management letter to physicians in case of allergic reactions was provided in 83.7 % (77/92) of patients. Oral food challenge (OFC) practices varied greatly concerning doses: most respondents used several doses (52/92, 56.5 %) during the same day (33/52, 63.4 %). Eleven responders (12.0 %) used the same protocol as for an IgE-mediated food allergy. CONCLUSION Our survey showed that practices of FPIES management in France are generally aligned with the international consensus guidelines. There are still pending issues to be standardised, such as the emergency kit prescription and its contents, as well as OFC management strategies. This work confirms the need for continuous training of physicians regarding FPIES management. Further guidelines are needed to improve standardisation of FPIES management.
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Affiliation(s)
- Amélie Perriere
- Sorbonne Université, Paediatric Nutrition and Gastroenterology Department, APHP-Trousseau Hospital, 26 avenue du Dr Arnold Netter, 75012 Paris, France.
| | - Karine Garcette
- Sorbonne Université, Paediatric Nutrition and Gastroenterology Department, APHP-Trousseau Hospital, 26 avenue du Dr Arnold Netter, 75012 Paris, France; CMSEA, 11 rue Froment, 75011 Paris, France.
| | - Nicolas Kalach
- Paediatric Department, Saint Antoine Paediatric Clinic, Saint Vincent de Paul Hospital, Catholic University of Lille, boulevard de Belfort, BP 387, 59020 Lille Cedex, France.
| | - Patrick Tounian
- Sorbonne Université, Paediatric Nutrition and Gastroenterology Department, APHP-Trousseau Hospital, 26 avenue du Dr Arnold Netter, 75012 Paris, France.
| | - Anaïs Lemoine
- Sorbonne Université, Paediatric Nutrition and Gastroenterology Department, APHP-Trousseau Hospital, 26 avenue du Dr Arnold Netter, 75012 Paris, France.
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Zubeldia‐Varela E, Blanco‐Pérez F, Barker‐Tejeda TC, Rojo D, Villaseñor A, Islam J, Gonzalez‐Menendez I, Laiño J, Krause M, Steigerwald H, Martella M, Quintanilla‐Martinez L, Yu P, Barbas C, Vieths S, Nochi T, Barber D, Toda M, Pérez‐Gordo M. The impact of high-IgE levels on metabolome and microbiome in experimental allergic enteritis. Allergy 2024; 79:3430-3447. [PMID: 38932655 PMCID: PMC11657046 DOI: 10.1111/all.16202] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/13/2022] [Revised: 04/03/2024] [Accepted: 05/08/2024] [Indexed: 06/28/2024]
Abstract
BACKGROUND The pathological mechanism of the gastrointestinal forms of food allergies is less understood in comparison to other clinical phenotypes, such as asthma and anaphylaxis Importantly, high-IgE levels are a poor prognostic factor in gastrointestinal allergies. METHODS This study investigated how high-IgE levels influence the development of intestinal inflammation and the metabolome in allergic enteritis (AE), using IgE knock-in (IgEki) mice expressing high levels of IgE. In addition, correlation of the altered metabolome with gut microbiome was analysed. RESULTS Ovalbumin-sensitized and egg-white diet-fed (OVA/EW) BALB/c WT mice developed moderate AE, whereas OVA/EW IgEki mice induced more aggravated intestinal inflammation with enhanced eosinophil accumulation. Untargeted metabolomics detected the increased levels of N-tau-methylhistamine and 2,3-butanediol, and reduced levels of butyric acid in faeces and/or sera of OVA/EW IgEki mice, which was accompanied with reduced Clostridium and increased Lactobacillus at the genus level. Non-sensitized and egg-white diet-fed (NC/EW) WT mice did not exhibit any signs of AE, whereas NC/EW IgEki mice developed marginal degrees of AE. Compared to NC/EW WT mice, enhanced levels of lysophospholipids, sphinganine and sphingosine were detected in serum and faecal samples of NC/EW IgEki mice. In addition, several associations of altered metabolome with gut microbiome-for example Akkermansia with lysophosphatidylserine-were detected. CONCLUSIONS Our results suggest that high-IgE levels alter intestinal and systemic levels of endogenous and microbiota-associated metabolites in experimental AE. This study contributes to deepening the knowledge of molecular mechanisms for the development of AE and provides clues to advance diagnostic and therapeutic strategies of allergic diseases.
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Affiliation(s)
- Elisa Zubeldia‐Varela
- Institute of Applied Molecular Medicine (IMMA), Department of Basic Medical Sciences, Facultad de MedicinaUniversidad San Pablo‐CEU, CEU UniversitiesMadridSpain
- Centre for Metabolomics and Bioanalysis (CEMBIO), Department of Chemistry and Biochemistry, Facultad de FarmaciaUniversidad San Pablo‐CEU, CEU UniversitiesMadridSpain
| | - Frank Blanco‐Pérez
- Molecular AllergologyPaul‐Ehrlich‐Institut, Federal Institute for Vaccines and BiomedicinesLangenGermany
| | - Tomás Clive Barker‐Tejeda
- Institute of Applied Molecular Medicine (IMMA), Department of Basic Medical Sciences, Facultad de MedicinaUniversidad San Pablo‐CEU, CEU UniversitiesMadridSpain
- Centre for Metabolomics and Bioanalysis (CEMBIO), Department of Chemistry and Biochemistry, Facultad de FarmaciaUniversidad San Pablo‐CEU, CEU UniversitiesMadridSpain
| | - David Rojo
- Centre for Metabolomics and Bioanalysis (CEMBIO), Department of Chemistry and Biochemistry, Facultad de FarmaciaUniversidad San Pablo‐CEU, CEU UniversitiesMadridSpain
| | - Alma Villaseñor
- Institute of Applied Molecular Medicine (IMMA), Department of Basic Medical Sciences, Facultad de MedicinaUniversidad San Pablo‐CEU, CEU UniversitiesMadridSpain
- Centre for Metabolomics and Bioanalysis (CEMBIO), Department of Chemistry and Biochemistry, Facultad de FarmaciaUniversidad San Pablo‐CEU, CEU UniversitiesMadridSpain
| | - Jahidul Islam
- Laboratory of Animal Functional Morphology, Graduate School of Agricultural ScienceTohoku UniversitySendaiJapan
| | - Irene Gonzalez‐Menendez
- Cluster of Excellence iFIT (EXC 2180) ‘Image Guided and Functionally Instructed Tumor Therapies’TübingenGermany
- Institute of Pathology and Neuropathology and Comprehensive Cancer Center TuebingenEberhard Karls UniversityTübingenGermany
| | - Jonathan Laiño
- Molecular AllergologyPaul‐Ehrlich‐Institut, Federal Institute for Vaccines and BiomedicinesLangenGermany
| | - Maren Krause
- Molecular AllergologyPaul‐Ehrlich‐Institut, Federal Institute for Vaccines and BiomedicinesLangenGermany
| | - Hanna Steigerwald
- Molecular AllergologyPaul‐Ehrlich‐Institut, Federal Institute for Vaccines and BiomedicinesLangenGermany
| | - Manuela Martella
- Institute of Pathology and Neuropathology and Comprehensive Cancer Center TuebingenEberhard Karls UniversityTübingenGermany
| | - Leticia Quintanilla‐Martinez
- Cluster of Excellence iFIT (EXC 2180) ‘Image Guided and Functionally Instructed Tumor Therapies’TübingenGermany
- Institute of Pathology and Neuropathology and Comprehensive Cancer Center TuebingenEberhard Karls UniversityTübingenGermany
| | - Philipp Yu
- Institute for ImmunologyPhilipps‐Universität MarburgMarburgGermany
| | - Coral Barbas
- Centre for Metabolomics and Bioanalysis (CEMBIO), Department of Chemistry and Biochemistry, Facultad de FarmaciaUniversidad San Pablo‐CEU, CEU UniversitiesMadridSpain
| | - Stefan Vieths
- Molecular AllergologyPaul‐Ehrlich‐Institut, Federal Institute for Vaccines and BiomedicinesLangenGermany
| | - Tomonori Nochi
- Laboratory of Animal Functional Morphology, Graduate School of Agricultural ScienceTohoku UniversitySendaiJapan
| | - Domingo Barber
- Institute of Applied Molecular Medicine (IMMA), Department of Basic Medical Sciences, Facultad de MedicinaUniversidad San Pablo‐CEU, CEU UniversitiesMadridSpain
| | - Masako Toda
- Molecular AllergologyPaul‐Ehrlich‐Institut, Federal Institute for Vaccines and BiomedicinesLangenGermany
- Laboratory of Food and Biomolecular Science, Graduate School of Agricultural ScienceTohoku UniversitySendaiJapan
| | - Marina Pérez‐Gordo
- Institute of Applied Molecular Medicine (IMMA), Department of Basic Medical Sciences, Facultad de MedicinaUniversidad San Pablo‐CEU, CEU UniversitiesMadridSpain
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Argiz L, Valsami-Fokianos M, Arasi S, Barni S, Boscia S, Bracaglia G, Bracamonte T, Carballeira I, Dinardo G, Echeverria L, Garcia E, Garcia-Magan C, Gomez-Rial J, Gonzalez-Delgado P, Fiocchi A, Garriga T, Ibrahim T, Infante S, Machinena A, Mangone G, Mori F, Moure JD, O'Valle V, Pascal M, Pecora V, Prieto A, Quevedo S, Salas A, Vazquez-Cortes S, Vila L, Martinon-Torres F, Gomez-Carballa A, Boyle RJ, Vazquez-Ortiz M. Clinical-Hematological Changes and Predictors of Severity in Acute Food Protein-Induced Enterocolitis Syndrome Reactions at Oral Food Challenge: A Multicenter Observational Study. THE JOURNAL OF ALLERGY AND CLINICAL IMMUNOLOGY. IN PRACTICE 2024; 12:2454-2467.e8. [PMID: 38796100 DOI: 10.1016/j.jaip.2024.05.024] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 07/24/2023] [Revised: 04/26/2024] [Accepted: 05/14/2024] [Indexed: 05/28/2024]
Abstract
BACKGROUND Oral food challenge (OFC) is the criterion standard for diagnosis of acute food protein-induced enterocolitis syndrome (FPIES). No diagnostic/prognostic biomarkers are available, and OFC assessment criteria are not validated. OBJECTIVE To assess clinical-hematological changes and predictors of severity of FPIES reactions at OFC. METHODS This was an observational multicenter prospective study. Children aged 0 to 18 years diagnosed with acute FPIES were recruited at follow-up OFC in 12 tertiary centers in Spain and Italy. OFC outcomes (as positive/negative/inconclusive and mild/moderate/severe) were assessed on the basis of published "2017 FPIES Consensus" criteria. Clinical characteristics were recorded, and full blood cell count was done at baseline, reaction onset, and 4 hours later. Regression analysis was performed to assess predictors of severe reactions at OFC. RESULTS A total of 81 children had positive OFC (mild in 11% [9 of 81], moderate in 61% [49 of 81], and severe in 28% [23 of 81]). Increase in neutrophils and reduction in eosinophils, basophils, and lymphocytes were observed (P < .05). OFC was inconclusive in 19 cases despite objective signs or neutrophilia. Regression analysis showed that a 2-day OFC protocol where only 25% of an age-appropriate portion is given on day 1 (not sex, age, culprit food, cumulative dose, and previous reaction severity) was associated with reduced odds of severe reaction compared with giving multiple doses in a single day. CONCLUSIONS Distinct hematological changes may help support FPIES diagnosis. Current OFC assessment criteria may not capture the broad spectrum of acute FPIES presentations. This 2-day protocol may be associated with a reduced risk of severe reactions. Future work should aim to develop safer OFC and non-OFC diagnostics for FPIES.
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Affiliation(s)
- L Argiz
- Department of Allergy, Clínica Universidad de Navarra, Pamplona, Spain; RICORS Red De Enfermedades Inflamatorias (REI) - RD21/0002/0028, Madrid, Spain
| | - M Valsami-Fokianos
- National Heart and Lung Institute, Imperial College London, London, United Kingdom
| | - S Arasi
- Allergy Unit, Department of Pediatric Medicine, Bambino Gesù Children's Research Hospital, IRCCS, Rome, Italy
| | - S Barni
- Allergy Unit, Meyer Children's Hospital IRCCS, Florence, Italy
| | - S Boscia
- Division of Immunology, Meyer Children's Hospital IRCCS, Florence, Italy; Department of Health Sciences, University of Florence, Florence, Italy
| | - G Bracaglia
- Laboratory Medicine, Bambino Gesù Children's Hospital IRCCS, Rome, Italy
| | - T Bracamonte
- Paediatric Allergy Section, Severo Ochoa University Hospital, Madrid, Spain
| | - I Carballeira
- Paediatric Allergy Section, Arquitecto Marcide Hospital, Coruña, Spain
| | - G Dinardo
- Department of Woman, Child and General and Specialized Surgery, University of Campania "Luigi Vanvitelli," Naples, Italy
| | - L Echeverria
- Paediatric Allergy Section, Severo Ochoa University Hospital, Madrid, Spain
| | - E Garcia
- Paediatric Allergy Section, Arquitecto Marcide Hospital, Coruña, Spain
| | - C Garcia-Magan
- Paediatrics Department, Hospital Clínico Universitario de Santiago de Compostela, Coruña, Spain
| | - J Gomez-Rial
- Genetics, Vaccines and Infections Research Group (GENVIP), Instituto de Investigación Sanitaria de Santiago (IDIS), University of Santiago de Compostela, Santiago de Compostela, Spain
| | | | - A Fiocchi
- Allergy Unit, Department of Pediatric Medicine, Bambino Gesù Children's Research Hospital, IRCCS, Rome, Italy
| | - T Garriga
- Paediatric Allergy Section, Vall D'Hebron University Hospital, Growth and Development Research Group, Vall d'Hebron Research Institute (VHIR), Barcelona, Spain
| | - T Ibrahim
- National Heart and Lung Institute, Imperial College London, London, United Kingdom; Allergy and Immunology Division, Hamad Medical Corporation, Doha, Qatar
| | - S Infante
- Pediatric Allergy Unit, Hospital General Universitario Gregorio Marañón, Gregorio Marañón Health Research Institute, IiSGM, Madrid, Spain
| | - A Machinena
- Pediatric Allergy and Clinical Immunology Department, Hospital Sant Joan de Déu, Barcelona, Spain
| | - G Mangone
- Division of Immunology, Meyer Children's Hospital IRCCS, Florence, Italy
| | - F Mori
- Allergy Unit, Meyer Children's Hospital IRCCS, Florence, Italy
| | - J D Moure
- Paediatrics Department, Hospital Clínico Universitario de Santiago de Compostela, Coruña, Spain
| | - V O'Valle
- Paediatric Allergy Section, Severo Ochoa University Hospital, Madrid, Spain
| | - M Pascal
- Immunology Department, CDB, Hospital Clínic de Barcelona, Barcelona, Spain; IDIBAPS, Universitat de Barcelona, Barcelona, Spain
| | - V Pecora
- Allergy Unit, Department of Pediatric Medicine, Bambino Gesù Children's Research Hospital, IRCCS, Rome, Italy
| | - A Prieto
- Paediatric Allergy Section, General University Hospital, Malaga, Spain
| | - S Quevedo
- Paediatric Allergy Section, Severo Ochoa University Hospital, Madrid, Spain
| | - A Salas
- Genetics, Vaccines and Infections Research Group (GENVIP), Instituto de Investigación Sanitaria de Santiago (IDIS), University of Santiago de Compostela, Santiago de Compostela, Spain; Unidade de Xenética, Instituto de Ciencias Forenses, Facultade de Medicina, Universidade de Santiago de Compostela, and GenPoB Research Group, Instituto de Investigación Sanitaria (IDIS), Hospital Clínico Universitario de Santiago de Compostela (SERGAS), Galicia, Spain
| | | | - L Vila
- Paediatric Allergy Section, Teresa Herrera Hospital, Coruña, Spain
| | - F Martinon-Torres
- Paediatrics Department, Hospital Clínico Universitario de Santiago de Compostela, Coruña, Spain; Genetics, Vaccines and Infections Research Group (GENVIP), Instituto de Investigación Sanitaria de Santiago (IDIS), University of Santiago de Compostela, Santiago de Compostela, Spain; Centro de Investigación Biomédica en Red de Enfermedades Respiratorias (CIBERES), Instituto de Salud Carlos III, Madrid, Spain
| | - A Gomez-Carballa
- Genetics, Vaccines and Infections Research Group (GENVIP), Instituto de Investigación Sanitaria de Santiago (IDIS), University of Santiago de Compostela, Santiago de Compostela, Spain; Unidade de Xenética, Instituto de Ciencias Forenses, Facultade de Medicina, Universidade de Santiago de Compostela, and GenPoB Research Group, Instituto de Investigación Sanitaria (IDIS), Hospital Clínico Universitario de Santiago de Compostela (SERGAS), Galicia, Spain
| | - R J Boyle
- Section of Inflammation, Repair and Development, National Heart and Lung Institute, Imperial College London, London, United Kingdom
| | - Marta Vazquez-Ortiz
- Section of Inflammation, Repair and Development, National Heart and Lung Institute, Imperial College London, London, United Kingdom.
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Vandenplas Y, Broekaert I, Domellöf M, Indrio F, Lapillonne A, Pienar C, Ribes-Koninckx C, Shamir R, Szajewska H, Thapar N, Thomassen RA, Verduci E, West C. An ESPGHAN Position Paper on the Diagnosis, Management, and Prevention of Cow's Milk Allergy. J Pediatr Gastroenterol Nutr 2024; 78:386-413. [PMID: 38374567 DOI: 10.1097/mpg.0000000000003897] [Citation(s) in RCA: 38] [Impact Index Per Article: 38.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/08/2022] [Accepted: 06/25/2023] [Indexed: 07/27/2023]
Abstract
A previous guideline on cow's milk allergy (CMA) developed by the European Society of Paediatric Gastroenterology, Hepatology and Nutrition (ESPGHAN) was published in 2012. This position paper provides an update on the diagnosis, treatment, and prevention of CMA with focus on gastrointestinal manifestations. All systematic reviews and meta-analyses regarding prevalence, pathophysiology, symptoms, and diagnosis of CMA published after the previous ESPGHAN document were considered. Medline was searched from inception until May 2022 for topics that were not covered in the previous document. After reaching consensus on the manuscript, statements were formulated and voted on each of them with a score between 0 and 9. A score of ≥6 was arbitrarily considered as agreement. Available evidence on the role of dietary practice in the prevention, diagnosis, and management of CMA was updated and recommendations formulated. CMA in exclusively breastfed infants exists, but is uncommon and suffers from over-diagnosis. CMA is also over-diagnosed in formula and mixed fed infants. Changes in stool characteristics, feeding aversion, or occasional spots of blood in stool are common and in general should not be considered as diagnostic of CMA, irrespective of preceding consumption of cow's milk. Over-diagnosis of CMA occurs much more frequently than under-diagnosis; both have potentially harmful consequences. Therefore, the necessity of a challenge test after a short diagnostic elimination diet of 2-4 weeks is recommended as the cornerstone of the diagnosis. This position paper contains sections on nutrition, growth, cost, and quality of life.
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Affiliation(s)
- Yvan Vandenplas
- Vrije Universiteit Brussel (VUB), UZ Brussel, KidZ Health Castle, Brussels, Belgium
| | - Ilse Broekaert
- Department of Paediatrics, Faculty of Medicine and University Hospital Cologne, University of Cologne, Cologne, Germany
| | - Magnus Domellöf
- Department of Clinical Sciences, Pediatrics, Umeå University, Umeå, Sweden
| | - Flavia Indrio
- Department of Medical and Surgical Science, University of Foggia, Foggia, Italy
| | - Alexandre Lapillonne
- Neonatal Intensive Care Unit, Necker-Enfants Malades Hospital, Paris University, Paris, France
- CNRC, Department of Pediatrics, Baylor College of Medicine, Houston, TX
| | - Corina Pienar
- Department of Pediatrics, "Victor Babes" University of Medicine and Pharmacy, Timisoara, Romania
| | - Carmen Ribes-Koninckx
- Gastroenterology and Hepatology & Instituto de Investigacion Sanitaria, La Fe University Hospital, Valencia, Spain
| | - Raanan Shamir
- Institute of Gastroenterology, Nutrition and Liver Diseases, Schneider Children's Medical Center, Lea and Arieh Pickel Chair for Pediatric Research, Sackler Faculty of Medicine, Tel-Aviv University, Tel-Aviv, Israel
| | - Hania Szajewska
- Department of Paediatrics, The Medical University of Warsaw, Warsaw, Poland
| | - Nikhil Thapar
- Gastroenterology, Hepatology and Liver Transplant, Queensland Children's Hospital, Brisbane, Australia
- School of Medicine, University of Queensland, Brisbane, Australia
- Woolworths Centre for Child Nutrition Research, Queensland University of Technology, Brisbane, Australia
- UCL Great Ormond Street Institute of Child Health, London, UK
| | - Rut Anne Thomassen
- Department of Paediatric Medicine, Division of Paediatric and Adolescent Medicine, Oslo University Hospital, Oslo, Norway
| | - Elvira Verduci
- Department of Paediatrics, Vittore Buzzi Children's Hospital, University of Milan, Milan, Italy
| | - Christina West
- Department of Clinical Sciences, Pediatrics, Umeå University, Umeå, Sweden
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7
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Nunes de Castilho Santos L. [Differential diagnosis in food allergy]. REVISTA ALERGIA MÉXICO 2023; 70:260-264. [PMID: 38506869 DOI: 10.29262/ram.v70i4.1312] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/01/2023] [Accepted: 10/29/2023] [Indexed: 03/21/2024] Open
Abstract
It is important to establish the differential diagnosis of food allergy with other disorders, for example: toxic reactions that occur in any person exposed to a sufficient amount of some allergen, and non-toxic reactions that depend on individual susceptibility (food allergy or intolerance). The differential diagnosis is decisive to establish the appropriate treatment. Food intolerance involves adverse reactions to foods without any immunological response involved, and commonly manifests with gastrointestinal symptoms (malaise, abdominal pain or diarrhea). Food allergy is an exaggerated reaction of the immune system, often mediated by IgE, that can trigger serious symptoms (hives, inflammation, respiratory distress, even anaphylaxis). The complex thing is because the symptoms sometimes overlap. To establish an accurate diagnosis, exhaustive clinical evaluation, laboratory tests and, in some cases, controlled provocation tests are required. It is important to understand these distinctions, because treatment and management vary significantly. Food intolerance involves the elimination or reduction of the food that triggers the allergic reaction and requires rigorous measures (complete avoidance of the allergen and availability of epinephrine in cases of severe reactions).
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Affiliation(s)
- Liziane Nunes de Castilho Santos
- Alergólogo e Inmunólogo; Responsable Técnico y Profesor del sector de Alergia e Inmunología del Instituto Nacional de Salud de la Mujer, del Niño y del Adolescente Fernandes Figueira IFF/Fiocruz,
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8
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Rojo Gutiérrez MI, Ballesteros González D, Ortiz Durán AK. [Non-IgE-mediated food allergy]. REVISTA ALERGIA MÉXICO 2023; 70:269-279. [PMID: 38506871 DOI: 10.29262/ram.v70i4.1338] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/01/2023] [Accepted: 10/29/2023] [Indexed: 03/21/2024] Open
Abstract
Food allergy is an immune response to proteins in food. It usually affects 8% of children and 2% of adults in Western countries. Non-IgE-mediated food allergy mainly affects the gastrointestinal tract. Gastrointestinal food allergies are classified, by their underlying pathogenesis, as: IgE-mediated, non-IgE-mediated, or mixed. The symptoms of patients with food protein-induced allergic proctocolitis originate from local inflammation of the distal colon, which causes hematochezia in neonates. It can affect the entire gastrointestinal tract and cause symptoms of intractable emesis, with subsequent metabolic disorders and hypovolemic shock. Food protein-induced enterocolitis syndrome is a non-IgE-mediated allergy that usually appears in childhood, with prolonged repetitive vomiting, starting 1 to 4 hours after ingestion of food. The manifestation in adults is usually triggered by the consumption of shellfish. Atopic diseases affect 40-60% of patients with food protein- induced enterocolitis syndrome, including 40-50% of those with food protein-induced enteropathy and proctocolitis. Probiotics (Lactobacillus GG) can alleviate the symptoms of allergic proctocolitis induced by food proteins, by altering the composition of the intestinal microbiota. Fecal microbiota transplantation (FMT) can change intestinal microecology efficiently compared to food or probiotics.
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Affiliation(s)
- María Isabel Rojo Gutiérrez
- Alergóloga e Inmunóloga clínica, Máster en Ciencias y Educación; Miembro de la Mesa Directiva de SLAAI; miembro activo del Colegio Mexicano de Inmunología Clínica y Alergia; Directora de Alergología en la Unidad Médica Zúrich, Ciudad de
| | - Diego Ballesteros González
- Médico Cirujano y Partero, Escuela Superior de Medicina, Instituto Politécnico Nacional; Alergia e inmunología clínica, Hospital Juárez de México
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9
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Mulé A, Prattico C, Al Ali A, Mulé P, Ben-Shoshan M. Diagnostic and Management Strategies of Food Protein-Induced Enterocolitis Syndrome: Current Perspectives. Pediatric Health Med Ther 2023; 14:337-345. [PMID: 37901587 PMCID: PMC10612481 DOI: 10.2147/phmt.s404779] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/18/2023] [Accepted: 10/13/2023] [Indexed: 10/31/2023] Open
Abstract
Food protein-induced enterocolitis syndrome (FPIES) is a form of non-IgE mediated food allergy that presents with delayed gastrointestinal symptoms after ingestion of the trigger food. The data regarding FPIES are sparse, despite being recognized as a distinct clinical entity. This narrative review presents the characteristics of this disorder in the pediatric population, as well-standard diagnostic and management protocols. FPIES can be classified into acute and chronic subtypes, and some cases may develop into an IgE-mediated allergy. Given that skin prick tests and specific IgE levels are negative in the majority of cases, diagnosis relies on clinical history and oral food challenges. Management involves elimination diets, assessment of tolerance through oral food challenges, and rehydration in the event of a reaction. Future research should focus on improving diagnostic methods, illustrating underlying pathogenesis and biomarkers, and assessing long-term natural history. Increased knowledge and awareness for FPIES are required.
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Affiliation(s)
- Angela Mulé
- Division of Allergy and Clinical Immunology, Department of Pediatrics, Montreal Children’s Hospital, McGill University Health Centre, Montreal, QC, Canada
| | - Catherine Prattico
- Division of Allergy and Clinical Immunology, Department of Pediatrics, Montreal Children’s Hospital, McGill University Health Centre, Montreal, QC, Canada
| | - Adnan Al Ali
- Division of Allergy and Clinical Immunology, Department of Pediatrics, Montreal Children’s Hospital, McGill University Health Centre, Montreal, QC, Canada
| | - Pasquale Mulé
- Division of Allergy and Clinical Immunology, Department of Pediatrics, Montreal Children’s Hospital, McGill University Health Centre, Montreal, QC, Canada
| | - Moshe Ben-Shoshan
- Division of Allergy and Clinical Immunology, Department of Pediatrics, Montreal Children’s Hospital, McGill University Health Centre, Montreal, QC, Canada
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10
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Hartono S, Zidan E, Sitaula P, Brooks JP. Pearls and pitfalls in food protein-induced enterocolitis syndrome (FPIES). Allergy Asthma Proc 2023; 44:368-373. [PMID: 37641223 PMCID: PMC10629436 DOI: 10.2500/aap.2023.44.230047] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 08/31/2023]
Abstract
Background: Food protein-induced enterocolitis syndrome (FPIES) is a rare, non-immunoglobulin E (IgE) mediated gastrointestinal food hypersensitivity. It is a clinical diagnosis commonly characterized by profuse vomiting 1 to 4 hours after ingestion of the triggering food(s). Objective: The objective was to increase awareness of FPIES and review the epidemiology, clinical presentation, pathogenesis, diagnosis, and management of FPIES. The lack of availability of a definite biomarker or diagnostic tool often leads to a delay in diagnosis. Methods: A literature search of salient articles that described case reports and case series of FPIES and their management were analyzed. Results: A case of FPIES with a literature review is presented with emphasis on clinical pearls and pitfalls. FPIES is a diagnosis of exclusion and the mainstay of treatment is avoidance of the trigger food(s) for at least 12-18 months from the last exposure. Conclusion: As FPIES is a non-IgE-mediated reaction, allergy testing via skin-prick test or blood tests to measure food IgE antibodies is not routinely recommended. Many children outgrow FPIES by 3-4 years of age. Supervised oral food challenge is recommended to assess acquisition of tolerance.
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Affiliation(s)
- Stella Hartono
- From the Laboratory of Allergic Diseases, National Institute of Allergy and Infectious Diseases, Bethesda, Maryland
| | - Elena Zidan
- Department of Internal Medicine, Bridgeport Hospital, Yale-New Haven Health System, Bridgeport, Connecticut, and
| | - Prasiksha Sitaula
- Division of Pediatric Allergy, Immunology, and Rheumatology, Columbia University Irving Medical Center, New York, New York
| | - Joel P Brooks
- Division of Pediatric Allergy, Immunology, and Rheumatology, Columbia University Irving Medical Center, New York, New York
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11
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Nishino M, Yanagida N, Sato S, Nagakura KI, Takahashi K, Ogura K, Ebisawa M. Prognosis of infantile food protein-induced enterocolitis syndrome to wheat: A case series. Pediatr Allergy Immunol 2023; 34:e13940. [PMID: 36974655 DOI: 10.1111/pai.13940] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/16/2022] [Revised: 02/25/2023] [Accepted: 03/03/2023] [Indexed: 03/29/2023]
Affiliation(s)
- Makoto Nishino
- Clinical Research Center for Allergy and Rheumatology, National Hospital Organization Sagamihara National Hospital, Kanagawa, Japan
- Department of Pediatrics, Ushiku Aiwa General Hospital, Ibaraki, Japan
| | - Noriyuki Yanagida
- Clinical Research Center for Allergy and Rheumatology, National Hospital Organization Sagamihara National Hospital, Kanagawa, Japan
- Department of Pediatrics, National Hospital Organization Sagamihara National Hospital, Kanagawa, Japan
| | - Sakura Sato
- Clinical Research Center for Allergy and Rheumatology, National Hospital Organization Sagamihara National Hospital, Kanagawa, Japan
| | - Ken-Ichi Nagakura
- Department of Pediatrics, National Hospital Organization Sagamihara National Hospital, Kanagawa, Japan
- Department of Pediatrics, Jikei University School of Medicine, Tokyo, Japan
| | - Kyohei Takahashi
- Department of Pediatrics, National Hospital Organization Sagamihara National Hospital, Kanagawa, Japan
| | - Kiyotake Ogura
- Department of Pediatrics, National Hospital Organization Sagamihara National Hospital, Kanagawa, Japan
| | - Motohiro Ebisawa
- Clinical Research Center for Allergy and Rheumatology, National Hospital Organization Sagamihara National Hospital, Kanagawa, Japan
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12
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Mathew M, Leeds S, Nowak-Węgrzyn A. Recent Update in Food Protein-Induced Enterocolitis Syndrome: Pathophysiology, Diagnosis, and Management. ALLERGY, ASTHMA & IMMUNOLOGY RESEARCH 2022; 14:587-603. [PMID: 36426394 PMCID: PMC9709682 DOI: 10.4168/aair.2022.14.6.587] [Citation(s) in RCA: 13] [Impact Index Per Article: 4.3] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Subscribe] [Scholar Register] [Received: 08/04/2022] [Revised: 09/29/2022] [Accepted: 10/07/2022] [Indexed: 11/26/2022]
Abstract
Food protein-induced enterocolitis syndrome (FPIES), though first reported in the 1970s, remains poorly understood and likely underdiagnosed. It is a non-immunoglobulin E (IgE)-mediated food allergy syndrome, most commonly identified in infancy and childhood. It can manifest as a constellation of symptoms following food ingestion, including repetitive and projectile emesis (1-4 hours), accompanied by pallor, lethargy, muscular hypotonia, and diarrhea (5-10 hours). In more severe reactions, significant leukocytosis with neutrophilia, thrombocytosis, metabolic derangements, methemoglobinemia, anemia, low albumin, and total protein may be present. Hypotension and ultimately hypovolemic distributive shock may occur in up to 15%-20% of cases. The diagnosis of FPIES is challenging and providers continue to face difficulties in management. This review article aims to highlight the most recent updates in epidemiology, natural history, pathophysiology, potential diagnostic markers, and guidelines for the management of FPIES.
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Affiliation(s)
- Mehr Mathew
- Department of Pediatrics, Yale School of Medicine, New Haven, CT, USA
| | - Stephanie Leeds
- Department of Pediatrics, Yale School of Medicine, New Haven, CT, USA
| | - Anna Nowak-Węgrzyn
- Department of Pediatrics, Hassenfeld Children's Hospital, NYU Grossman School of Medicine, New York, NY, USA
- Department of Pediatrics, Gastroenterology and Nutrition, Collegium Medicum, University of Warmia and Mazury, Olsztyn, Poland.
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13
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Hayano S, Natsume O, Yasuoka R, Katoh Y, Koda M. Predictors of initial oral food challenge outcome in food protein-induced enterocolitis syndrome. THE JOURNAL OF ALLERGY AND CLINICAL IMMUNOLOGY. GLOBAL 2022; 1:122-127. [PMID: 37781265 PMCID: PMC10509941 DOI: 10.1016/j.jacig.2022.05.004] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Subscribe] [Scholar Register] [Received: 12/16/2021] [Revised: 04/12/2022] [Accepted: 05/09/2022] [Indexed: 10/03/2023]
Abstract
Background There is a paucity of data on predictors of clinical history in oral food challenge (OFC) outcome for the initial diagnosis of food protein-induced enterocolitis syndrome (FPIES). Objective This study aimed to identify predictors for the diagnosis of FPIES. Methods The study included patients who underwent OFC to diagnose FPIES from 2010 to 2021. Patients with a positive OFC result were classified as belonging to the FPIES group, and those with negative OFC result within 120 days from the last symptomatic episode were classified as belonging to the no-allergy (NA) group. Background factors were analyzed in the groups. Results A total of 50 OFCs to 12 different foods were conducted in 50 patients. Of those 50 patients, 30 were classified as belonging to the FPIES group. No significant difference was observed between the FPIES and NA groups with respect to background factors, including the features of symptomatic episodes and examinations of immediate-type allergy. A history of asymptomatic ingestion was observed in 23 of 24 and 13 of 19 patients in the FPIES and NA groups, respectively; thus, it was significantly more common in patients with FPIES. The diagnostic rate of patients with fewer than 3 symptomatic episodes was 52%, and that of patients with 3 episodes or more was 75%, not considering a patient without available data. Conclusions A definite diagnosis of FPIES should be based on OFC, as there are no predictors for OFC positivity other than a history of asymptomatic ingestion. The absence of asymptomatic ingestion history was a negative predictor for the diagnosis of FPIES.
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Affiliation(s)
- Satoshi Hayano
- Allergic Disease Research Center, Chutoen General Medical Center, Kakegawa, Japan
- Department of Pediatrics, Chutoen General Medical Center, Kakegawa, Japan
| | - Osamu Natsume
- Department of Pediatrics, Chutoen General Medical Center, Kakegawa, Japan
- Department of Pediatrics, Hamamatsu University School of Medicine, Hamamatsu City, Japan
- Department of Pediatrics, Public Morimachi Hospital, Morimachi, Japan
| | - Ryuhei Yasuoka
- Department of Pediatrics, Hamamatsu University School of Medicine, Hamamatsu City, Japan
| | - Yukiko Katoh
- Department of Pediatrics, Hamamatsu University School of Medicine, Hamamatsu City, Japan
| | - Masaki Koda
- Department of Pediatrics, Hamamatsu University School of Medicine, Hamamatsu City, Japan
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14
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Anvari S, Ruffner MA. Adult Food Protein-Induced Enterocolitis Syndrome. FRONTIERS IN ALLERGY 2022; 3:889879. [PMID: 35769585 PMCID: PMC9234874 DOI: 10.3389/falgy.2022.889879] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.7] [Reference Citation Analysis] [Abstract] [Grants] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/04/2022] [Accepted: 04/25/2022] [Indexed: 11/13/2022] Open
Abstract
Food protein-induced enterocolitis syndrome (FPIES) is a non-IgE, cell-mediated food allergy, commonly diagnosed in infants and young children. In recent years, new-onset adult FPIES has been recognized. The underlying pathogenic mechanism of FPIES has yet to be elucidated, thus disease-specific diagnostic biomarkers have yet to be determined and an oral food challenge (OFC) remains the gold-standard for the diagnosis. Pediatric patients with FPIES classically present with symptoms of delayed, repetitive vomiting approximately 1 to 4 hours following ingestion of a food allergen. However, adults with FPIES have been reported to have a different symptom profile and different food triggers compared to the pediatric FPIES population. The current FPIES diagnostic criteria may not be appropriate for the diagnosis of adult FPIES patients, thus an oral food challenge remains a diagnostic tool. This review provides an overview of the current literature on the clinical presentation, epidemiology, diagnosis, triggers and management of adult FPIES.
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Affiliation(s)
- Sara Anvari
- Division of Immunology, Allergy, and Retrovirology, Department of Pediatrics, Texas Children's Hospital, Baylor College of Medicine, Houston, TX, United States
- William T. Shearer Center for Human Immunobiology, Texas Children's Hospital, Houston, TX, United States
- *Correspondence: Sara Anvari
| | - Melanie A. Ruffner
- Division of Allergy and Immunology, Children's Hospital of Philadelphia, Philadelphia, PA, United States
- Department of Pediatrics, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA, United States
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15
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Elghoudi A, Narchi H. Food allergy in children-the current status and the way forward. World J Clin Pediatr 2022; 11:253-269. [PMID: 35663006 PMCID: PMC9134150 DOI: 10.5409/wjcp.v11.i3.253] [Citation(s) in RCA: 25] [Impact Index Per Article: 8.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/06/2021] [Revised: 07/16/2021] [Accepted: 03/16/2022] [Indexed: 02/06/2023] Open
Abstract
Food allergy in children is a major health concern, and its prevalence is rising. It is often over-diagnosed by parents, resulting occasionally in unnecessary exclusion of some important food. It also causes stress, anxiety, and even depression in parents and affects the family's quality of life. Current diagnostic tests are useful when interpreted in the context of the clinical history, although cross-sensitivity and inability to predict the severity of the allergic reactions remain major limitations. Although the oral food challenge is the current gold standard for making the diagnosis, it is only available to a small number of patients because of its requirement in time and medical personnel. New diagnostic methods have recently emerged, such as the Component Resolved Diagnostics and the Basophil Activation Test, but their use is still limited, and the latter lacks standardisation. Currently, there is no definite treatment available to induce life-long natural tolerance and cure for food allergy. Presently available treatments only aim to decrease the occurrence of anaphylaxis by enabling the child to tolerate small amounts of the offending food, usually taken by accident. New evidence supports the early introduction of the allergenic food to infants to decrease the incidence of food allergy. If standardised and widely implemented, this may result in decreasing the prevalence of food allergy.
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Affiliation(s)
- Ahmed Elghoudi
- Paediatric Department, Sheikh Khalifa Medical City, Abu Dhabi NA, Abu Dhabi, United Arab Emirates
- College of Medicine and Health Sciences, United Arab Emirates University, Alain, Abu Dhabi, United Arab Emirates
| | - Hassib Narchi
- College of Medicine and Health Sciences, United Arab Emirates University, Alain, Abu Dhabi, United Arab Emirates
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16
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Zhang S, Sicherer S, Berin MC, Agyemang A. Pathophysiology of Non-IgE-Mediated Food Allergy. Immunotargets Ther 2022; 10:431-446. [PMID: 35004389 PMCID: PMC8721028 DOI: 10.2147/itt.s284821] [Citation(s) in RCA: 37] [Impact Index Per Article: 12.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/02/2021] [Accepted: 12/24/2021] [Indexed: 12/12/2022] Open
Abstract
Non-IgE-mediated food allergies are a group of disorders characterized by subacute or chronic inflammatory processes in the gut. Unlike IgE mediated food allergies that may result in multi-organ system anaphylaxis, the non-IgE mediated food allergies primarily affect the gastrointestinal tract. This review outlines the clinical manifestations, epidemiology, pathophysiology, and management of non-IgE-mediated food allergies. An updated literature search of selected non-IgE-mediated food allergies was conducted for this review using PubMed database to the current year (2021). Reviewed disorders include food protein-induced enterocolitis syndrome (FPIES), food-protein enteropathy (FPE), food protein-induced allergic proctocolitis (FPIAP), and eosinophilic gastrointestinal disorders (EGIDs) such as eosinophilic esophagitis (EoE). While extensive gains have been made in understanding FPIES, FPIAP, FPE, and EoE, more information is needed on the pathophysiology of these food allergies. Similarities among them include involvement of innate immunity, T-lymphocyte processes, alteration of the intestinal lumen at the cellular level with the appearance of inflammatory cells and associated histologic changes, and specific cytokine profiles suggesting food-specific, T-cell, and immune-mediated responses. While FPIES and FPIAP typically resolve in early childhood, EGIDs typically do not. Emerging new therapies for EoE offer promise of additional treatment options. Further studies identifying the immunopathogenesis, associated biomarkers, and mechanisms of tolerance are needed to inform prevention, diagnosis and management.
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Affiliation(s)
- Shouling Zhang
- Department of Pediatrics, Division of Allergy and Immunology, Icahn School of Medicine at Mount Sinai, Kravis Children's Hospital, The Elliot and Roslyn Jaffe Food Allergy Institute, New York, NY, USA
| | - Scott Sicherer
- Department of Pediatrics, Division of Allergy and Immunology, Icahn School of Medicine at Mount Sinai, Kravis Children's Hospital, The Elliot and Roslyn Jaffe Food Allergy Institute, New York, NY, USA
| | - M Cecilia Berin
- Department of Pediatrics, Division of Allergy and Immunology, Icahn School of Medicine at Mount Sinai, Kravis Children's Hospital, The Elliot and Roslyn Jaffe Food Allergy Institute, New York, NY, USA
| | - Amanda Agyemang
- Department of Pediatrics, Division of Allergy and Immunology, Icahn School of Medicine at Mount Sinai, Kravis Children's Hospital, The Elliot and Roslyn Jaffe Food Allergy Institute, New York, NY, USA
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17
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Bulsa K, Standowicz M, Baryła-Pankiewicz E, Czaja-Bulsa G. Chronic Milk-Dependent Food Protein-Induced Enterocolitis Syndrome in Children from West Pomerania Region. Nutrients 2021; 13:nu13114137. [PMID: 34836392 PMCID: PMC8617799 DOI: 10.3390/nu13114137] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/03/2021] [Revised: 11/15/2021] [Accepted: 11/17/2021] [Indexed: 12/13/2022] Open
Abstract
Characteristics of chronic milk-dependent food protein-induced enterocolitis syndrome (FPIES) in children from the region of Western Pomerania were studied. Prospectively, 55 children were diagnosed at a median of 2.2 months. The open food challenges (OFC), morphologies, milk-specific IgE (sIgE) (FEIA method, CAP system), and skin prick tests (SPTs) were examined. Vomiting and diarrhea escalated gradually but quickly led to growth retardation. Of the infants, 49% had BMI < 10 c, 20% BMI < 3 c; 25% had anemia, and 15% had hypoalbuminemia. During the OFCs we observed acute symptoms that appeared after 2-3 h: vomiting diarrhea and pallor. A total of 42% children required intravenous hydration. Casein hydrolysates or amino acids formulae (20%) were used in treatment. In 25% of children, SPT and milk sIgE were found, in 18%-other food SPTs, and in 14% allergy to other foods. A transition to IgE-dependent milk allergy was seen in 3 children. In the twelfth month of life, 62% of children had tolerance to milk, and in the twenty-fifth month-87%. Conclusions. Chronic milk-dependent FPIES resolves in most children. By the age of 2 children are at risk of multiple food sensitization, and those who have milk sIgE are at risk to transition to IgE-mediated milk allergy. Every OFC needs to be supervised due to possible severe reactions.
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Affiliation(s)
| | | | | | - Grażyna Czaja-Bulsa
- Chair and Department of Paediatrics and Paediatric Nursing, Pomeranian Medical University, 70-204 Szczecin, Poland
- Correspondence: ; Tel.: +48-91-480-09-51; Fax: +48-91-880-61-46
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18
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Zubeldia-Varela E, Barker-Tejeda TC, Blanco-Pérez F, Infante S, Zubeldia JM, Pérez-Gordo M. Non-IgE-Mediated Gastrointestinal Food Protein-Induced Allergic Disorders. Clinical Perspectives and Analytical Approaches. Foods 2021; 10:foods10112662. [PMID: 34828942 PMCID: PMC8623505 DOI: 10.3390/foods10112662] [Citation(s) in RCA: 12] [Impact Index Per Article: 3.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/13/2021] [Accepted: 10/27/2021] [Indexed: 12/21/2022] Open
Abstract
Non-IgE-mediated gastrointestinal food allergy (non-IgE-GI-FA) is the name given to a series of pathologies whose main entities are food protein-induced allergic proctocolitis (FPIAP), food protein-induced enteropathy (FPE), and food protein-induced enterocolitis syndrome (FPIES). These are more uncommon than IgE-mediated food allergies, their mechanisms remain largely unknown, and their diagnosis is mainly done by clinical history, due to the lack of specific biomarkers. In this review, we present the latest advances found in the literature about clinical aspects, the current diagnosis, and treatment options of non-IgE-GI-FAs. We discuss the use of animal models, the analysis of gut microbiota, omics techniques, and fecal proteins with a focus on understanding the pathophysiological mechanisms of these pathologies and obtaining possible diagnostic and/or prognostic biomarkers. Finally, we discuss the unmet needs that researchers should tackle to advance in the knowledge of these barely explored pathologies.
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Affiliation(s)
- Elisa Zubeldia-Varela
- Department of Basic Medical Sciences, Facultad de Medicina, Institute of Applied Molecular Medicine (IMMA), Universidad San Pablo-CEU, CEU Universities, ARADyAL, 28660 Madrid, Spain; (E.Z.-V.); (T.C.B.-T.)
- Centre for Metabolomics and Bioanalysis (CEMBIO), Department of Chemistry and Biochemistry, Facultad de Farmacia, Universidad San Pablo-CEU, CEU Universities, Urbanización Montepríncipe, Boadilla del Monte, 28660 Madrid, Spain
| | - Tomás Clive Barker-Tejeda
- Department of Basic Medical Sciences, Facultad de Medicina, Institute of Applied Molecular Medicine (IMMA), Universidad San Pablo-CEU, CEU Universities, ARADyAL, 28660 Madrid, Spain; (E.Z.-V.); (T.C.B.-T.)
- Centre for Metabolomics and Bioanalysis (CEMBIO), Department of Chemistry and Biochemistry, Facultad de Farmacia, Universidad San Pablo-CEU, CEU Universities, Urbanización Montepríncipe, Boadilla del Monte, 28660 Madrid, Spain
| | - Frank Blanco-Pérez
- VPr1 Research Group “Molecular Allergology”, Paul-Ehrlich-Institut, Federal Institute for Vaccines and Biomedicines, 63225 Langen, Germany;
| | - Sonsoles Infante
- Allergy Paediatric Unit, Allergy Service, Hospital General Universitario Gregorio Marañón, 28007 Madrid, Spain; (S.I.); (J.M.Z.)
- Gregorio Marañón Health Research Institute (IiSGM), 28007 Madrid, Spain
| | - José M. Zubeldia
- Allergy Paediatric Unit, Allergy Service, Hospital General Universitario Gregorio Marañón, 28007 Madrid, Spain; (S.I.); (J.M.Z.)
- Gregorio Marañón Health Research Institute (IiSGM), 28007 Madrid, Spain
- Rare Diseases Networking Biomedical Research Centre (CIBERER, U-761), Carlos III Institute of Health, 28029 Madrid, Spain
| | - Marina Pérez-Gordo
- Department of Basic Medical Sciences, Facultad de Medicina, Institute of Applied Molecular Medicine (IMMA), Universidad San Pablo-CEU, CEU Universities, ARADyAL, 28660 Madrid, Spain; (E.Z.-V.); (T.C.B.-T.)
- Correspondence: ; Tel.: +34-91-372-4700 (ext. 14675)
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19
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Boyer J, Sgambelluri L, Yuan Q. Association of Antibiotic Usage with Food Protein-Induced Enterocolitis Syndrome Development from a Caregiver's Survey. JPGN REPORTS 2021; 2:e132. [PMID: 35373193 PMCID: PMC8966620 DOI: 10.1097/pg9.0000000000000132] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Subscribe] [Scholar Register] [Received: 07/02/2021] [Accepted: 08/28/2021] [Indexed: 05/20/2023]
Abstract
Background Food protein-induced enterocolitis syndrome (FPIES) is a frequently misdiagnosed, serious, non-IgE mediated food allergy, and the precise mechanism of disease is unknown. Acute FPIES typically presents with repetitive, profuse vomiting approximately 1-4 hours post ingestion of a food trigger. Chronic FPIES is considered less common and less well characterized. Objective We aimed to better describe FPIES and identify factors that may influence FPIES development through use of a self-reported, caregiver's survey. Methods FPIES and allergy-free infant caregivers completed a survey regarding lifestyle factors that may influence allergy acquisition such as: antibiotic usage and delivery mode. FPIES caregivers reported symptoms, number of food triggers, type of FPIES, and symptoms from breastmilk ingestion. FPIES infants were compared to allergy-free infants to identify factors potentially associated with FPIES. Results Infant and prenatal maternal antibiotic usage was higher in FPIES infants compared to allergy-free infants (43.8% vs 20.6% and 48.8% vs 23.57%, respectively, p< 0.05). When compared to infants with ACUTE FPIES alone, infants described as BOTH acute and chronic FPIES reported earlier onset of symptoms, more non-specific symptoms and symptoms triggered by breast milk, more antibiotic exposure, and more food triggers (p< 0.05). Conclusion Antibiotic usage was significantly higher in FPIES infants when compared to allergy-free infants. Work is needed to elucidate the role of antibiotic usage in the etiology of FPIES. Infants reported to have BOTH acute and chronic FPIES were significantly different from infants with ACUTE FPIES alone highlighting the need to more closely examine these different subtypes of FPIES.
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Affiliation(s)
| | | | - Qian Yuan
- MassGeneral Hospital for Children, Boston, MA
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20
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Kim YI, Joo JY, Jung YH, Choi CW, Kim BI, Yang HR. Differentiation of food protein-induced enterocolitis syndrome misleading to necrotizing enterocolitis. Ann Allergy Asthma Immunol 2021; 128:193-198. [PMID: 34626783 DOI: 10.1016/j.anai.2021.09.024] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/24/2021] [Revised: 09/10/2021] [Accepted: 09/30/2021] [Indexed: 11/19/2022]
Abstract
BACKGROUND Food protein-induced enterocolitis syndrome (FPIES) is a rare non-immunoglobulin E-mediated food allergy with necrotizing enterocolitis (NEC)-like symptoms which requires differential diagnosis as treatments differ. OBJECTIVE To evaluate the clinical, laboratory, and radiologic findings that differentiate FPIES from NEC in preterm and term infants. METHODS Clinical features, comorbidities, and laboratory and radiologic findings of neonates with presumed NEC were reviewed retrospectively and compared between FPIES and NEC in preterm and term infants who were admitted to the neonatal intensive care unit at Seoul National University Bundang Hospital between May 2003 and February 2020. RESULTS A total of 10 of 150 (6.7%) preterm and 17 of 38 (44.7%) term infants with presumed NEC were confirmed to have FPIES; the remainder had NEC by modified Bell's criteria. Demographics and comorbidities were similar between these groups. Symptoms such as hematochezia, shock, leukocytosis, peripheral eosinophilia, and feeding of extensively hydrolyzed milk formula or elemental formula after discharge were significantly different between the 2 groups in term infants (P <.05), but not in preterm infants. On abdominal ultrasonography, pneumatosis intestinalis was more common among preterm FPIES (44.4%) than NEC cases (21.6%) (P =.04). Among the preterm infants, 4 FPIES (40%) and 25 NEC (17.9%) cases required surgery (P =.10). CONCLUSION Differentiating FPIES in neonates suspected of having NEC is important as dietary elimination of the triggering milk protein can be recommended instead of prolonged fasting and antibiotic treatment, which are indicated for NEC, in both term and preterm infants.
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Affiliation(s)
- You Ie Kim
- Department of Pediatrics, Seoul National University Bundang Hospital, Seongnam, Republic of Korea
| | - Jung Yeon Joo
- Department of Pediatrics, Seoul National University Bundang Hospital, Seongnam, Republic of Korea
| | - Young Hwa Jung
- Department of Pediatrics, Seoul National University Bundang Hospital, Seongnam, Republic of Korea
| | - Chang Won Choi
- Department of Pediatrics, Seoul National University Bundang Hospital, Seongnam, Republic of Korea; Department of Pediatrics, Seoul National University College of Medicine, Seoul, Republic of Korea
| | - Beyoung Il Kim
- Department of Pediatrics, Seoul National University Bundang Hospital, Seongnam, Republic of Korea; Department of Pediatrics, Seoul National University College of Medicine, Seoul, Republic of Korea
| | - Hye Ran Yang
- Department of Pediatrics, Seoul National University Bundang Hospital, Seongnam, Republic of Korea; Department of Pediatrics, Seoul National University College of Medicine, Seoul, Republic of Korea.
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21
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Di Chio T, Sokollik C, Peroni DG, Hart L, Simonetti G, Righini-Grunder F, Borrelli O. Nutritional Aspects of Pediatric Gastrointestinal Diseases. Nutrients 2021; 13:nu13062109. [PMID: 34205445 PMCID: PMC8235230 DOI: 10.3390/nu13062109] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/03/2021] [Revised: 06/09/2021] [Accepted: 06/15/2021] [Indexed: 12/16/2022] Open
Abstract
In the last decade, the role of nutritional management in pediatric gastrointestinal diseases has gained increasing popularity. Disease-specific diets have been introduced as conventional treatments by international guidelines. Patients tend to more willingly accept food-based therapies than drugs because of their relatively “harmless” nature. Apart from a diet’s therapeutic role, nutritional support is crucial in maintaining growth and improving clinical outcomes in pediatric patients. Despite the absence of classical “side effects”, however, it should be emphasized that any dietary modification might have negative consequences on children’s growth and development. Hence, expert supervision is always advised, in order to support adequate nutritional requirements. Unfortunately, the media provide an inaccurate perception of the role of diet for gastrointestinal diseases, leading to misconceptions by patients or their caregivers that tends to overestimate the beneficial role of diets and underestimate the potential adverse effects. Moreover, not only patients, but also healthcare professionals, have a number of misconceptions about the nutritional benefits of diet modification on gastrointestinal diseases. The aim of this review is to highlight the role of diet in pediatric gastrointestinal diseases, to detect misconceptions and to give a practical guide for physicians on the basis of current scientific evidence.
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Affiliation(s)
- Teresa Di Chio
- Pediatric Institute of Southern Switzerland, Ospedale Regionale di Bellinzona e Valli, Via Ospedale 12, 6500 Bellinzona, Switzerland;
- Correspondence: (T.D.C.); (C.S.); (F.R.-G.); (O.B.)
| | - Christiane Sokollik
- Division of Pediatric Gastroenterology, Hepatology and Nutrition, Children’s Hospital, Inselspital, University of Bern, 3010 Bern, Switzerland
- Correspondence: (T.D.C.); (C.S.); (F.R.-G.); (O.B.)
| | - Diego G. Peroni
- Department of Clinical and Experimental Medicine, Section of Pediatrics, University of Pisa, 56126 Pisa, Italy;
| | - Lara Hart
- Division of Pediatric Gastroenterology, Hepatology and Nutrition, McMaster University, Hamilton, ON L8N 3Z5, Canada;
| | - Giacomo Simonetti
- Pediatric Institute of Southern Switzerland, Ospedale Regionale di Bellinzona e Valli, Via Ospedale 12, 6500 Bellinzona, Switzerland;
- Università della Svizzera Italiana, 6900 Lugano, Switzerland
| | - Franziska Righini-Grunder
- Division of Pediatric Gastroenterology, Hepatology and Nutrition, Lucerne Children’s Hospital, Cantonal Hospital Lucerne, 6000 Lucerne, Switzerland
- Correspondence: (T.D.C.); (C.S.); (F.R.-G.); (O.B.)
| | - Osvaldo Borrelli
- Division of Neurogastroenterology and Motility, Department of Pediatric Gastroenterology, University College London (UCL) Institute of Child Health and Great Ormond Street, London WC1N 3JH, UK
- Correspondence: (T.D.C.); (C.S.); (F.R.-G.); (O.B.)
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22
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Food protein-induced enterocolitis syndrome: epidemiology and comorbidities. Curr Opin Allergy Clin Immunol 2021; 20:168-174. [PMID: 31977448 DOI: 10.1097/aci.0000000000000615] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/15/2022]
Abstract
PURPOSE OF REVIEW First described in the mid 20th century, it was just in the last decade that diagnostic and treatment guidelines for food protein-induced enterocolitis syndrome (FPIES) were established. Awareness of the diagnosis is improving, and epidemiologic data are emerging. RECENT FINDINGS Recent studies suggest that FPIES may affect as many as 0.5% of children worldwide. FPIES in adults is usually triggered by seafood and may be more common than previously thought. Many patients with FPIES have other allergic disorders. SUMMARY With refined diagnostic criteria and improved awareness, FPIES is now diagnosed with increasing frequency, and epidemiologic data are emerging. FPIES appears to be increasing in prevalence, and the frequent association with other allergic disorders suggests a shared predisposition or immune mechanism that remains to be elucidated.
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23
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Nowak-Wegrzyn A, Berin MC, Mehr S. Food Protein-Induced Enterocolitis Syndrome. THE JOURNAL OF ALLERGY AND CLINICAL IMMUNOLOGY-IN PRACTICE 2021; 8:24-35. [PMID: 31950904 DOI: 10.1016/j.jaip.2019.08.020] [Citation(s) in RCA: 82] [Impact Index Per Article: 20.5] [Reference Citation Analysis] [Abstract] [Key Words] [Subscribe] [Scholar Register] [Received: 07/03/2019] [Revised: 08/14/2019] [Accepted: 08/21/2019] [Indexed: 01/01/2023]
Abstract
Food protein-induced enterocolitis syndrome (FPIES) is a non-IgE-mediated food allergy that manifests with projectile, repetitive emesis that can be followed by diarrhea and may be accompanied by lethargy, hypotonia, hypothermia, hypotension, and metabolic derangements. FPIES usually starts in infancy although onset at older ages is being increasingly recognized. FPIES is not rare, with the cumulative incidence of FPIES in infants estimated to be 0.015% to 0.7%, whereas the population prevalence in the US infants was 0.51%. FPIES diagnosis is challenging and might be missed because of later (1-4 hours) onset of symptoms after food ingestion, lack of typical allergic skin and respiratory symptoms, and food triggers that are perceived to be hypoallergenic. Diagnosis is based on the recognition of symptoms because there are no biomarkers of FPIES. The pathophysiology remains obscure although activation of the innate immune compartment has been detected. Management relies of avoidance of food triggers, treatment of accidental exposures, and periodic re-evaluations with supervised oral food challenges to monitor for resolution. There are no strategies to accelerate development of tolerance in FPIES. Here we review the most important current concepts in epidemiology, pathophysiology, diagnosis, and management of FPIES.
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Affiliation(s)
- Anna Nowak-Wegrzyn
- Department of Pediatrics, New York University Langone Health, New York, NY; Department of Pediatrics, Gastroenterology and Nutrition, Collegium Medicum, University of Warmia and Mazury, Olsztyn, Poland.
| | - M Cecilia Berin
- Precision Immunology Institute, Jaffe Food Allergy Institute, Icahn School of Medicine at Mount Sinai, New York, NY
| | - Sam Mehr
- Department of Allergy and Immunology, Royal Children's Hospital, Melbourne, VIC, Australia
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24
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Barni S, Giovannini M, Mori F. Epidemiology of non-IgE-mediated food allergies: what can we learn from that? Curr Opin Allergy Clin Immunol 2021; 21:188-194. [PMID: 33394702 DOI: 10.1097/aci.0000000000000721] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/18/2022]
Abstract
PURPOSE OF REVIEW To underline the main characteristics of the non-Immunoglobulin E (IgE)-mediated food allergies (food protein-induced allergic proctocolitis food protein-induced enteropathy and food protein-induced enterocolitis syndrome ), which are common diseases in primary care and in allergy and gastroenterology specialty practices evaluating children. RECENT FINDINGS Non-IgE-mediated food allergies comprise a spectrum of diseases with peculiar features affecting infants and young children. The most prominent features of these diseases are symptoms that affect mainly the gastrointestinal tract. SUMMARY It is of paramount importance to provide the clinicians with the tools for non-IgE-mediated food allergy recognition in clinical practice to avoid the misdiagnosis with unnecessary laboratory tests and detrimental treatments.
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Affiliation(s)
- Simona Barni
- Allergy Unit, Department of Pediatrics, Meyer Children's University Hospital, Florence, Italy
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25
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Infante S, Pérez-Pallisé E, Skrabski F, Cabrera-Freitag P, Morales-Cabeza C, Fuentes-Aparicio V, Alvarez-Perea A, Zubeldia JM. Poor prognosis of food protein-induced enterocolitis syndrome to fish. Pediatr Allergy Immunol 2021; 32:560-565. [PMID: 33336438 DOI: 10.1111/pai.13430] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/04/2020] [Revised: 12/07/2020] [Accepted: 12/11/2020] [Indexed: 11/29/2022]
Abstract
BACKGROUND Fish is the most common causative food of food protein-induced enterocolitis syndrome (FPIES) in Southern Europe. In children with FPIES, the development of tolerance varies according to the culprit food and specifically fish seems to have a poorer prognosis than other solid foods. We sought to evaluate the fish-FPIES resolution rate in children. METHODS A descriptive retrospective analysis of children with fish-FPIES, followed during the last 20 years, was performed. The offending fish, age and symptoms at onset, the coexistence of atopic diseases and FPIES to other foods were registered. All the children included had undergone an oral food challenge (OFC) with the offending fish. We recorded those children that overcame their fish-FPIES and those that did not outgrow the disease. RESULTS Seventy children were enrolled in this study (median age: 9 yo; IQR 6.4-13.8). Forty-two (60%) achieved tolerance to the offending fish with a median age of 4 years (IQR: 3-5). Among children ≤5 yo (n = 40), 35 (87.5%) developed tolerance; among 6-8yo (n = 14), 40% developed tolerance; and only 12.5% among those ≥9 yo (n = 16) developed tolerance. Twenty-eight children did not outgrow the disease (median age: 8.9 yo; IQR: 9-13.8). We did not find any statistical differences regarding the offending fish, presence of single vs multiple fish-FPIES, symptoms at the beginning, coexistence of other atopic diseases or the coexistence of other FPIES, between the children who overcame the disease and those who did not. CONCLUSION One in five children with FPIES to fish will not overcome the disease during childhood.
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Affiliation(s)
- Sonsoles Infante
- Paediatric Allergy Unit, Hospital General Universitario Gregorio Marañón, Madrid, Spain.,Gregorio Marañón Health Research Institute (IiSGM), Madrid, Spain
| | | | - Filip Skrabski
- Paediatric Allergy Unit, Hospital General Universitario Gregorio Marañón, Madrid, Spain
| | - Paula Cabrera-Freitag
- Paediatric Allergy Unit, Hospital General Universitario Gregorio Marañón, Madrid, Spain.,Gregorio Marañón Health Research Institute (IiSGM), Madrid, Spain
| | | | - Victoria Fuentes-Aparicio
- Paediatric Allergy Unit, Hospital General Universitario Gregorio Marañón, Madrid, Spain.,Gregorio Marañón Health Research Institute (IiSGM), Madrid, Spain
| | - Alberto Alvarez-Perea
- Paediatric Allergy Unit, Hospital General Universitario Gregorio Marañón, Madrid, Spain.,Gregorio Marañón Health Research Institute (IiSGM), Madrid, Spain
| | - José M Zubeldia
- Paediatric Allergy Unit, Hospital General Universitario Gregorio Marañón, Madrid, Spain.,Gregorio Marañón Health Research Institute (IiSGM), Madrid, Spain.,Biomedical Research Network on Rare Diseases (CIBERER)-U761, Madrid, Spain
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26
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Groetch M, Baker MG, Durban R, Meyer R, Venter C, Muraro A. The practical dietary management of food protein-induced enterocolitis syndrome. Ann Allergy Asthma Immunol 2021; 127:28-35. [PMID: 33757808 DOI: 10.1016/j.anai.2021.03.007] [Citation(s) in RCA: 13] [Impact Index Per Article: 3.3] [Reference Citation Analysis] [Abstract] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/14/2020] [Revised: 03/03/2021] [Accepted: 03/16/2021] [Indexed: 12/13/2022]
Abstract
OBJECTIVE Food protein-induced enterocolitis syndrome (FPIES) is a non-immunoglobulin E-mediated food allergy with potential risk of malnutrition related to the early onset of disease, frequent avoidance of cow's milk, and the possibility of multiple food triggers. This publication is aimed at providing an evidence-based, practical approach to the dietary management of FPIES. DATA SOURCES This is a narrative review summarizing information from national and international guidelines, retrospective studies, population studies, review articles, case reports, and case series to evaluate for nutritional risk and develop guidance for risk reduction in children with FPIES. STUDY SELECTIONS We have included retrospective clinical cohort studies, population-based studies, case reports, and case studies. We did not exclude any studies identified owing to the small number of studies addressing the nutritional management of individuals with FPIES. RESULTS Children with FPIES are at risk of malnutrition owing to suboptimal oral intake, limited food choices, and knowledge deficits related to feeding. In particular, children with 3 or more FPIES triggers seem to be at increased risk for poor weight gain and developing food aversion. Caregivers of children with FPIES also report a high degree of psychosocial burden. CONCLUSION Appropriate dietary management entails the following 3 essential components: supporting normal growth and development, avoidance of allergens, and advancement of complementary foods. Education to avoid the trigger food and assisting caregivers in creating an individualized, well-designed complementary feeding plan to meet the infant's nutritional needs for optimal growth and development are essential management strategies.
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Affiliation(s)
- Marion Groetch
- Division of Pediatric Allergy and Immunology, Department of Pediatrics, Icahn School of Medicine at Mount Sinai, New York, New York.
| | - Mary Grace Baker
- Division of Pediatric Allergy and Immunology, Department of Pediatrics, Icahn School of Medicine at Mount Sinai, New York, New York
| | - Raquel Durban
- Asthma & Allergy Specialists, Charlotte, North Carolina
| | - Rosan Meyer
- Department of Paediatrics, Imperial College London, London, United Kingdom
| | - Carina Venter
- Section of Pediatric Allergy and Clinical Immunology, Children's Hospital Colorado, University of Colorado School of Medicine, Aurora, Colorado
| | - Antonella Muraro
- Food Allergy Referral Centre, Department of Woman and Child Health, Padua University Hospital, Padua, Italy
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27
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Food protein-induced enterocolitis syndrome oral food challenge: Time for a change? Ann Allergy Asthma Immunol 2021; 126:506-515. [PMID: 33662509 DOI: 10.1016/j.anai.2021.02.022] [Citation(s) in RCA: 23] [Impact Index Per Article: 5.8] [Reference Citation Analysis] [Abstract] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/06/2020] [Revised: 02/11/2021] [Accepted: 02/22/2021] [Indexed: 11/20/2022]
Abstract
OBJECTIVE Food protein-induced enterocolitis syndrome (FPIES) is typically diagnosed based on a characteristic clinical history; however, an oral food challenge (OFC) may be necessary to confirm the diagnosis or evaluate for the development of tolerance. FPIES OFC methods vary globally, and there is no universally agreed upon protocol. The objective of this review is to summarize reported FPIES OFC approaches and consider unmet needs in diagnosing and managing FPIES. DATA SOURCES PubMed database was searched using the keywords food protein-induced enterocolitis syndrome, oral food challenge, cow milk allergy, food allergy, non-immunoglobulin E-mediated food allergy and FPIES. STUDY SELECTIONS Primary and review articles were selected based on relevance to the diagnosis of FPIES and the FPIES OFC. RESULTS We reviewed the history of FPIES and the evolution and variations in the FPIES OFC. A summary of current literature suggests that most patients with FPIES will react with 25% to 33% of a standard serving of the challenged food, there is little benefit to offering a divided dose challenge unless there is suspicion of specific immunoglobulin E to the food being challenged, reactions typically appear within 1 to 4 hours of ingestion, and reactions during OFC rarely result in emergency department or intensive care unit admission. CONCLUSION International standardization in the FPIES OFC approach is necessary with particular attention to specific dose administration across challenged foods, timing between the patient's reaction and offered OFC to verify tolerance, patient safety considerations before the OFC, and identification of characteristics that would indicate home reintroduction is appropriate.
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28
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Cianferoni A. Food protein-induced enterocolitis syndrome epidemiology. Ann Allergy Asthma Immunol 2021; 126:469-477. [PMID: 33607250 DOI: 10.1016/j.anai.2021.02.006] [Citation(s) in RCA: 24] [Impact Index Per Article: 6.0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/17/2020] [Revised: 02/05/2021] [Accepted: 02/10/2021] [Indexed: 12/12/2022]
Abstract
OBJECTIVE Food protein-induced enterocolitis syndrome (FPIES) is a condition with heterogeneous features (ie, age at presentation, severity, food triggers, comorbidities) and is not as rare as initially believed. In the last few years, the first population-based epidemiologic study, few prospective birth cohort evaluating FPIES prevalence, and several larger (>100 patients) studies have been published, making epidemiologic estimation more reliable. In this review, we report on the available data on the epidemiology of FPIES. DATA SOURCES PubMed review using the following words: FPIES, epidemiology, and prevalence. STUDY SELECTIONS The review focused on the population-based epidemiologic study, few prospective birth cohort evaluating FPIES prevalence, and several larger (>100 patients) studies. RESULTS We identified 8 population or cohort studies. CONCLUSION FPIES is not rare in both children and adults and may affect as many as 900,000 people in the United States alone. Most children and adult with FPIES seem to react to 1 to 2 foods; however, they may need further diet restriction owing to high level of comorbidity with immunoglobulin E-mediated food allergies and eosinophilic esophagitis. Globally, cow's milk, rice/oat, and seafood seem to be the most common triggers.
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Affiliation(s)
- Antonella Cianferoni
- Division of Allergy and Immunology, The Children's Hospital of Philadelphia and Perelman School of Medicine, University of Pennsylvania, Philadelphia, Pennsylvania.
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29
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Watanabe Y, Sakai H, Nihei M, Miura K, Kumaki S. Early tolerance acquisition in hen's egg yolk-associated food protein-induced enterocolitis syndrome. THE JOURNAL OF ALLERGY AND CLINICAL IMMUNOLOGY. IN PRACTICE 2021; 9:2120-2122.e2. [PMID: 33440257 DOI: 10.1016/j.jaip.2020.12.053] [Citation(s) in RCA: 9] [Impact Index Per Article: 2.3] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 09/19/2020] [Revised: 12/18/2020] [Accepted: 12/18/2020] [Indexed: 11/17/2022]
Affiliation(s)
- Yohei Watanabe
- Department of Pediatrics, Sendai Medical Center, Sendai, Japan.
| | - Hideyuki Sakai
- Department of Pediatrics, Sendai Medical Center, Sendai, Japan
| | - Masato Nihei
- Department of Pediatrics, Sendai Medical Center, Sendai, Japan; Department of Allergy, Miyagi Children's Hospital, Sendai, Japan
| | - Katsushi Miura
- Department of Allergy, Miyagi Children's Hospital, Sendai, Japan
| | - Satoru Kumaki
- Department of Pediatrics, Sendai Medical Center, Sendai, Japan
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30
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Barni S, Vazquez-Ortiz M, Giovannini M, Liccioli G, Sarti L, Cianferoni A, Mori F. 'Diagnosing food protein-induced enterocolitis syndrome'. Clin Exp Allergy 2021; 51:14-28. [PMID: 33089888 DOI: 10.1111/cea.13767] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/27/2020] [Revised: 10/03/2020] [Accepted: 10/17/2020] [Indexed: 12/13/2022]
Abstract
Food protein-induced enterocolitis syndrome is still a mysterious disease, pathogenically poorly characterized, although the first FPIES case has been described in 1967. Mainly, food protein-induced enterocolitis syndrome diagnosis is based on clinical history. The oral food challenge remains the gold standard to confirm the diagnosis, especially in particular situations. Although there are no diagnostic laboratory or imaging tests which are specific for diagnosis, they could, however, sometimes be helpful to rule out clinical conditions which are similar to food protein-induced enterocolitis syndrome reactions. The purpose of this review is to define the clinical features of FPIES and to summarize the current available tools for the diagnosis of FPIES. This review is intended to be a practical guide for the clinician facing a patient with food protein-induced enterocolitis syndrome avoiding delayed diagnosis with unnecessary laboratory tests and detrimental treatments. Moreover, it highlights the unmet needs in diagnosis that require urgent attention from the scientific community to improve the management of patients with FPIES.
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Affiliation(s)
- Simona Barni
- Allergy Unit, Meyer Children's University Hospital, Florence, Italy
| | - Marta Vazquez-Ortiz
- Section of Inflammation, Repair and Development, National Heart and Lung Institute, Imperial College London, London, UK
| | | | - Giulia Liccioli
- Allergy Unit, Meyer Children's University Hospital, Florence, Italy
| | - Lucrezia Sarti
- Allergy Unit, Meyer Children's University Hospital, Florence, Italy
| | - Antonella Cianferoni
- Children's Hospital of Philadelphia, University of Pennsylvania Medical School, Philadelphia, PA, USA
| | - Francesca Mori
- Allergy Unit, Meyer Children's University Hospital, Florence, Italy
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31
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Ocak M, Akarsu A, Sahiner UM, Soyer O, Sekerel BE. Phenotypes and natural history of food protein-induced enterocolitis syndrome in the east Mediterranean region. Allergy Asthma Proc 2020; 41:420-427. [PMID: 33109307 DOI: 10.2500/aap.2020.41.200078] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/08/2023]
Abstract
Background: Food protein-induced enterocolitis syndrome (FPIES) is a rare non-IgE mediated food allergy. Objective: To delineate the differences in the spectrum of culprit foods, remission patterns, and predictors among varying cultures. Methods: We reviewed demographics, culprit foods, outcomes, and predictors in 81 children with a diagnosis of FPIES who were followed up between 2015 and 2020. Results: Eighty-one patients (55.6% boys) were enrolled, including 72 with acute FPIES and 9 with chronic FPIES. Hen's egg was the most common culprit food (36.6%), followed by fish (26.9%), and cow's milk (21.5%). Interestingly, cow's milk was significantly prevalent in chronic FPIES cases (p = 0.006). The most common clinical symptoms were vomiting (100%), pallor (63.4%), and lethargy (55.9%). Emergency department visits were noted in 39 patients (41.9%), of whom 37 (39.8%) were treated with intravenous (IV) fluid. The subjects were followed up for a median (interquartile range) of 19.4 months (12.3-41.2 months), and 26 subjects (32.1%) achieved tolerance. The median (interquartile range) age at tolerance was 2.5 years (2.1-3.2 years). With regard to the culprit foods, hen's egg was observed more frequently in the subjects with resolved FPIES cases (p = 0.008), whereas fish FPIES cases were high in the persistent group (p = 0.001). IgE sensitization of the culprit food was found to be an independent risk factor for the persistence of FPIES (odds ratio 4.855 [95% confidence interval, 1.131-20.844]; p = 0.034). Conclusion: In our cohort, unlike other published series, hen's egg and fish were the two most common culprit foods. Fish differed from other culprit foods, with significantly delayed onset and persistence, and may create a model that allows for the understanding of the disease.
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Affiliation(s)
- Melike Ocak
- From the Department of Pediatric Allergy, Faculty of Medicine, Hacettepe University, Ankara, Turkey
| | - Ayşegül Akarsu
- From the Department of Pediatric Allergy, Faculty of Medicine, Hacettepe University, Ankara, Turkey
| | - Umit Murat Sahiner
- From the Department of Pediatric Allergy, Faculty of Medicine, Hacettepe University, Ankara, Turkey
| | - Ozge Soyer
- From the Department of Pediatric Allergy, Faculty of Medicine, Hacettepe University, Ankara, Turkey
| | - Bulent Enis Sekerel
- From the Department of Pediatric Allergy, Faculty of Medicine, Hacettepe University, Ankara, Turkey
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Makita E, Kuroda S, Sato H, Itabashi K, Kawano A, Matsuura M, Sugiyama Y, Sugawara D, Maruyama A, Ichihashi K. Comparison of methemoglobin levels in food protein-induced enterocolitis syndrome and other gastrointestinal diseases in neonates. Allergol Immunopathol (Madr) 2020; 48:490-495. [PMID: 32451130 DOI: 10.1016/j.aller.2020.01.007] [Citation(s) in RCA: 8] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/02/2019] [Revised: 01/12/2020] [Accepted: 01/29/2020] [Indexed: 12/23/2022]
Abstract
INTRODUCTION AND OBJECTIVES Methemoglobinemia has been reported to be associated with severe food protein-induced enterocolitis syndrome (FPIES). However, no reports have evaluated methemoglobin (MHb) levels in FPIES without symptomatic methemoglobinemia or the usefulness of MHb measurement for the diagnostic prediction of FPIES. To evaluate the MHb levels of patients with neonatal-onset FPIES and determine whether MHb levels are higher in FPIES than in other gastrointestinal diseases. PATIENTS AND METHODS Eleven neonates with severe acute FPIES (FPIES group) and 139 neonates with other gastrointestinal diseases (non-FPIES group) were included in this study. Patient characteristics, symptoms, and venous blood test values (MHb, pH, HCO3-, and C-reactive protein) were evaluated. RESULTS The median age at onset was 16 days vs. 1 day; males comprised 64% vs. 46%, the median gestational age was 38 weeks vs. 38 weeks, the median birth weight was 2710g vs. 2880g, and the median hospitalization duration was 31 days vs. 6 days for the FPIES vs. non-FPIES groups, respectively. MHb (%) was higher in the FPIES group than in the non-FPIES group [median (range), 1.1 (0.6-10.9) and 0.6 (0.3-1.2), respectively, p<0.001]. There were no differences in terms of pH, HCO3-, and C-reactive protein (p>0.05). In the receiver operating characteristic analysis for FPIES diagnosis based on MHb (%), the area under the curve was 0.885, specificity was 97.1%, and sensitivity was 72.7% at a MHb cutoff of 1.0. CONCLUSION High MHb levels may help diagnose severe acute FPIES in neonates, but careful evaluation is needed.
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Affiliation(s)
- Eishi Makita
- Department of Pediatrics, Saitama Medical Center, Jichi Medical University, Saitama 330-8503, Japan.
| | - Sae Kuroda
- Department of Pediatrics, Saitama Medical Center, Jichi Medical University, Saitama 330-8503, Japan; Department of Pediatrics, Saitama Red Cross Hospital, Saitama 330-8553, Japan
| | - Hiroaki Sato
- Department of Pediatrics, Saitama Medical Center, Jichi Medical University, Saitama 330-8503, Japan
| | - Kae Itabashi
- Department of Pediatrics, Saitama Medical Center, Jichi Medical University, Saitama 330-8503, Japan
| | - Atsuko Kawano
- Department of Pediatrics, Saitama Medical Center, Jichi Medical University, Saitama 330-8503, Japan
| | - Misa Matsuura
- Department of Pediatrics, Saitama Medical Center, Jichi Medical University, Saitama 330-8503, Japan
| | - Yohei Sugiyama
- Department of Pediatrics, Saitama Medical Center, Jichi Medical University, Saitama 330-8503, Japan
| | - Daisuke Sugawara
- Department of Pediatrics, Saitama Medical Center, Jichi Medical University, Saitama 330-8503, Japan
| | - Asami Maruyama
- Department of Pediatrics, Saitama Medical Center, Jichi Medical University, Saitama 330-8503, Japan
| | - Ko Ichihashi
- Department of Pediatrics, Saitama Medical Center, Jichi Medical University, Saitama 330-8503, Japan
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Mastrorilli C, Santoro A, Procaccianti M, Pagliaro G, Caffarelli C. New insights into food protein-induced enterocolitis in children. Minerva Pediatr 2020; 72:416-423. [PMID: 32686925 DOI: 10.23736/s0026-4946.20.05976-9] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/08/2022]
Abstract
Food protein-induced enterocolitis syndrome (FPIES) represents a non-IgE-mediated food allergic disorder with delayed gastrointestinal symptoms that may evolve in a medical emergency. Clinically, FPIES can be distinguished into acute and chronic phenotypes. FPIES is mainly diagnosed in infancy however the onset at older ages is being progressively described. The pathogenetic mechanism underlying FPIES remains mainly unexplained, but an alteration of food-specific T-cell response has been proposed. The diagnosis of FPIES is primarily clinical, since there are not available specific biomarkers. Oral food challenge (OFC) is the gold standard for diagnosing FPIES or excluding the onset of tolerance to the triggering food. Management of FPIES includes an acute phase treatment and a maintenance therapy with the strict food avoidance until challenge, in order to prevent new attacks and avoid nutritional alterations. Acute management requires hydration that can be performed orally or intravenously according to clinical status. Long-term management of FPIES is based on the avoidance of the culprit food(s) and supervised introduction of other high-risk foods if never taken before among infants before 12 months of age. There is a compelling need of future achievements in FPIES research for the definition of underlying disease pathogenesis and potential therapeutic point of care.
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Affiliation(s)
- Carla Mastrorilli
- Unit of Pediatric Allergy and Pulmonology, Department of Pediatrics and Emergency, Consorziale-Policlinico University Hospital, Pediatric Hospital Giovanni XXIII, Bari, Italy -
| | - Angelica Santoro
- Department of Medicine and Surgery, Pediatric Clinic, University of Parma, Parma, Italy
| | - Michela Procaccianti
- Department of Medicine and Surgery, Pediatric Clinic, University of Parma, Parma, Italy
| | - Giuseppe Pagliaro
- Pediatric Unit, Department of Obstetrics, Gynecology and Pediatrics, Arcispedale Santa Maria Nuova-IRCCS, Reggio Emilia, Italy
| | - Carlo Caffarelli
- Department of Medicine and Surgery, Pediatric Clinic, University of Parma, Parma, Italy
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Labrosse R, Graham F, Caubet JC. Non-IgE-Mediated Gastrointestinal Food Allergies in Children: An Update. Nutrients 2020; 12:nu12072086. [PMID: 32674427 PMCID: PMC7400851 DOI: 10.3390/nu12072086] [Citation(s) in RCA: 71] [Impact Index Per Article: 14.2] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/11/2020] [Revised: 06/26/2020] [Accepted: 07/02/2020] [Indexed: 02/07/2023] Open
Abstract
Non-immunoglobulin E-mediated gastrointestinal food allergic disorders (non-IgE-GI-FA) include food protein-induced enterocolitis syndrome (FPIES), food protein-induced enteropathy (FPE) and food protein-induced allergic proctocolitis (FPIAP), which present with symptoms of variable severity, affecting the gastrointestinal tract in response to specific dietary antigens. The diagnosis of non-IgE-GI-FA is made clinically, and relies on a constellation of typical symptoms that improve upon removal of the culprit food. When possible, food reintroduction should be attempted, with the documentation of symptoms relapse to establish a conclusive diagnosis. Management includes dietary avoidance, nutritional counselling, and supportive measures in the case of accidental exposure. The prognosis is generally favorable, with the majority of cases resolved before school age. Serial follow-up to establish whether the acquisition of tolerance has occurred is therefore essential in order to avoid unnecessary food restriction and potential consequent nutritional deficiencies. The purpose of this review is to delineate the distinctive clinical features of non-IgE-mediated food allergies presenting with gastrointestinal symptomatology, to summarize our current understanding of the pathogenesis driving these diseases, to discuss recent findings, and to address currents gaps in the knowledge, to guide future management opportunities.
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Affiliation(s)
- Roxane Labrosse
- Division of Hematology-Oncology, Department of Pediatrics, Boston Children’s Hospital, Boston, MA 02115, USA;
- Division of Allergy and Immunology, Department of Pediatrics, CHU Sainte-Justine, University of Montreal, Montreal, QC H3T 1C5, Canada;
| | - François Graham
- Division of Allergy and Immunology, Department of Pediatrics, CHU Sainte-Justine, University of Montreal, Montreal, QC H3T 1C5, Canada;
- Division of Allergy and Immunology, Department of Medicine, Centre Hospitalier de l’Universite de Montreal (CHUM), University of Montreal, Montreal, QC H2X 3E4, Canada
| | - Jean-Christoph Caubet
- Pediatric Allergy Unit, Department of Woman, Child and Adolescent, University Hospitals of Geneva, 1205 Geneva, Switzerland
- Correspondence:
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Unique features of non-IgE-mediated gastrointestinal food allergy during infancy in Japan. Curr Opin Allergy Clin Immunol 2020; 20:299-304. [DOI: 10.1097/aci.0000000000000642] [Citation(s) in RCA: 8] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/13/2022]
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Abstract
Food allergies, defined as an immune response to food proteins, affect as many as 8% of young children and 2% of adults in western countries, and their prevalence appears to be rising like all allergic diseases. In addition to well-recognized urticaria and anaphylaxis triggered by IgE antibody- mediated immune responses, there is an increasing recognition of cell-mediated disorders, such as eosinophilic esophagitis and food protein-induced enterocolitis. Non-IgE-Mediated gastrointestinal food allergies are a heterogeneous group of food allergies in which there is an immune reaction against food but the primary pathogenesis is not a production of IgE and activation of mast cells and basophils. Those diseases tend to affect mainly the gastrointestinal tract and can present as acute (FPIES) or chronic reaction, such as Eosinophilic Esophagitis (EoE), Food Protein-Induced Allergic Proctocolitis (FPIAP). The role of food allergy in Non-EoE gastrointestinal Eosinophilic disorders (Non- EoE EGID) is poorly understood. In some diseases like EoE, T cell seems to play a major role in initiating the immunological reaction against food, however, in FPIES and FPIAP, the mechanism of sensitization is not clear. Diagnosis requires food challenges and/or endoscopies in most of the patients, as there are no validated biomarkers that can be used for monitoring or diagnosis of Non-IgE mediated food allergies. The treatment of Non-IgE food allergy is dependent on diet (FPIES, and EoE) and/or use of drugs (i.e. steroids, PPI) in EoE and Non-EoE EGID. Non-IgE mediated food allergies are being being investigated.
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Affiliation(s)
- Antonella Cianferoni
- Division of Allergy and Immunology, Department of Pediatrics, The Children's Hospital of Philadelphia, University of Pennsylvania School of Medicine, Philadelphia, PA 19104, United States
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Infante S, Cabrera-Freitag P, Morales-Cabeza C, Alvarez-Perea A. Geographical Variations in Food Protein-Induced Enterocolitis Syndrome. CURRENT TREATMENT OPTIONS IN ALLERGY 2019. [DOI: 10.1007/s40521-019-00234-x] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/27/2022]
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Douros K, Tsabouri S, Feketea G, Grammeniatis V, Koliofoti EG, Papadopoulos M, Sardeli O, Triga M, Priftis KN. Retrospective study identified fish and milk as the main culprits in cases of food protein-induced enterocolitis syndrome. Acta Paediatr 2019; 108:1901-1904. [PMID: 30883926 DOI: 10.1111/apa.14779] [Citation(s) in RCA: 17] [Impact Index Per Article: 2.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/20/2019] [Revised: 03/06/2019] [Accepted: 03/12/2019] [Indexed: 02/07/2023]
Abstract
AIM Food protein-induced enterocolitis syndrome (FPIES) is a non-immunoglobulin E (IgE)-mediated food allergy, which is confined to the gastrointestinal tract and occurs most frequently in the first year of life. Our aim was to examine the clinical features, causative agents and outcomes of Greek children with FPIES. METHODS This was a five-year (2013-2017) retrospective study, based on chart reviews of 78 children with FPIES from six Greek paediatric allergy centres. RESULTS Only five children needed an oral food challenge (OFC) for diagnosis, but 54 children (69%) had OFCs to monitor tolerance development. The most common problems were fish and milk, which affected affecting 42 (54%) and 25 (35%) of children, respectively. The median age of tolerance based on the results of the negative OFCs occurred by 34.0 (26.6-58.4) months. Fish and milk were tolerated by 24 (57%) and 13 (52%) of children by 43.8 and 24.3 months, respectively. Coexisting IgE sensitisation to the offending food was related to delayed tolerance. CONCLUSION Fish and milk were the most common food allergies in our series of Greek children with FPIES. Cases with IgE sensitisation to the food trigger took longer to resolve their allergies.
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Affiliation(s)
- Konstantinos Douros
- Allergology and Pulmonology Unit 3rd Paediatric Department Attikon Hospital National and Kapodistrian University of Athens Athens Greece
| | - Sophia Tsabouri
- Child Health Department University of Ioannina School of Medicine Ioannina Greece
| | | | - Vasilis Grammeniatis
- Allergology and Pulmonology Unit 3rd Paediatric Department Attikon Hospital National and Kapodistrian University of Athens Athens Greece
- Department of Paediatrics General Hospital of Ioannina Ioannina Greece
| | | | - Marios Papadopoulos
- Allergology and Pulmonology Unit 3rd Paediatric Department Attikon Hospital National and Kapodistrian University of Athens Athens Greece
- Iaso Children's Hospital Athens Greece
| | - Olympia Sardeli
- Allergology and Pulmonology Unit 3rd Paediatric Department Attikon Hospital National and Kapodistrian University of Athens Athens Greece
| | - Maria Triga
- Department of Paediatrics University General Hospital of Patras Patras Greece
| | - Kostas N. Priftis
- Allergology and Pulmonology Unit 3rd Paediatric Department Attikon Hospital National and Kapodistrian University of Athens Athens Greece
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Caubet J, Cianferoni A, Groetch M, Nowak‐Wegrzyn A. Food protein‐induced enterocolitis syndrome. Clin Exp Allergy 2019; 49:1178-1190. [DOI: 10.1111/cea.13415] [Citation(s) in RCA: 15] [Impact Index Per Article: 2.5] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/19/2018] [Revised: 05/03/2019] [Accepted: 05/03/2019] [Indexed: 12/21/2022]
Affiliation(s)
- Jean‐Christoph Caubet
- Geneva University Hospital Geneva Switzerland
- Division of Allergy and Immunology, Department of Pediatrics, Jaffe Food Allergy Institute, Icahn School of Medicine at Mount Sinai Kravis Children's Hospital New York New York
| | - Antonella Cianferoni
- Children's Hospital of Philadelphia University of Pennsylvania Medical School Philadelphia Pennsylvania
| | - Marion Groetch
- Division of Allergy and Immunology, Department of Pediatrics, Jaffe Food Allergy Institute, Icahn School of Medicine at Mount Sinai Kravis Children's Hospital New York New York
| | - Anna Nowak‐Wegrzyn
- Division of Allergy and Immunology, Department of Pediatrics, Jaffe Food Allergy Institute, Icahn School of Medicine at Mount Sinai Kravis Children's Hospital New York New York
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Xepapadaki P, Kitsioulis NA, Manousakis E, Manolaraki I, Douladiris N, Papadopoulos NG. Remission Patterns of Food Protein-Induced Enterocolitis Syndrome in a Greek Pediatric Population. Int Arch Allergy Immunol 2019; 180:113-119. [PMID: 31390649 DOI: 10.1159/000500860] [Citation(s) in RCA: 23] [Impact Index Per Article: 3.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/18/2019] [Accepted: 05/09/2019] [Indexed: 11/19/2022] Open
Abstract
BACKGROUND Data on the prevalence and clinical course of food protein-induced enterocolitis syndrome (FPIES) vary between populations and according to the culprit food. OBJECTIVE To evaluate the incidence, clinical characteristics, and remission patterns of FPIES among children in a Greek pediatric allergy referral center. METHODS We retrospectively studied children with acute FPIES. Data on age, sex, type of reaction, the implicated food, and oral food challenge (OFC) outcomes at baseline and upon reevaluation were analyzed. RESULTS Between October 2010 and March 2017, 72 (38 males) out of 15,114 subjects who had been referred to our department due to any reported allergic symptoms were diagnosed with acute FPIES. The most commonly implicated foods were cow's milk (CM) (45.8%), fish (34.7%), rice (9.7%), egg (6.9%), and chicken (2.8%). The mean age at diagnosis was 7.1/19.3/9.1/18.7/8.7 months for those with CM/fish/rice/egg/chicken FPIES, respectively. Sixty-nine OFCs were performed, of which 8 were diagnostic and 61 for tolerance evaluation. The type of culprit food was significantly associated with the outcome of the tolerance OFCs. OFCs to fish resulted positive at a significantly higher rate (12/22; 54.5%) than OFCs to CM (4/29; 13.7%), rice (1/5; 20%), egg (0/3; 0%), and chicken (0/2; 0%) (p = 0.01). The time period between diagnosis and tolerance acquisition was prolonged in the fish FPIES cases (74.8 months; 95% CI: 57.9-91.6) compared to that with other foods such as CM (20.7 months; 95% CI: 17.3-24.1), rice (31.8 months; 95% CI: 21.9-41.7), and egg (24.3 months; 95% CI: 10.7-37.9), as shown in a Kaplan-Meier analysis (log-rank, p < 0.001). When the fish FPIES children were assessed for tolerance, OFCs were significantly more often positive than in CM FPIES children (52 vs. 18.1%; p = 0.03), despite the fact that the children were challenged at an older age (fish: 70.4 months, 95% CI: 58.3-82.5, vs. CM: 26.57 months, 95% CI: 21.1-32, p < 0.001). CONCLUSIONS Acute FPIES had a low incidence in our population. CM and fish were the two most frequent elicitors. Significantly delayed presentation and prolonged remission was noted for FPIES caused by fish.
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Affiliation(s)
- Paraskevi Xepapadaki
- Allergy Department, 2nd Pediatric Clinic, National and Kapodistrian University of Athens, Athens, Greece,
| | | | - Emmanouil Manousakis
- Allergy Department, 2nd Pediatric Clinic, National and Kapodistrian University of Athens, Athens, Greece
| | - Ioanna Manolaraki
- Allergy Department, 2nd Pediatric Clinic, National and Kapodistrian University of Athens, Athens, Greece
| | - Nikolaos Douladiris
- Allergy Department, 2nd Pediatric Clinic, National and Kapodistrian University of Athens, Athens, Greece
| | - Nikolaos G Papadopoulos
- Allergy Department, 2nd Pediatric Clinic, National and Kapodistrian University of Athens, Athens, Greece.,Division of Infection, Immunity and Respiratory Medicine, University of Manchester, Manchester, United Kingdom
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Meyer R, Fox AT, Chebar Lozinsky A, Michaelis LJ, Shah N. Non-IgE-mediated gastrointestinal allergies-Do they have a place in a new model of the Allergic March. Pediatr Allergy Immunol 2019; 30:149-158. [PMID: 30403301 DOI: 10.1111/pai.13000] [Citation(s) in RCA: 18] [Impact Index Per Article: 3.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/05/2018] [Revised: 10/08/2018] [Accepted: 10/23/2018] [Indexed: 12/18/2022]
Abstract
The rise in food allergy has been described as the "second wave" of the allergy epidemic, with some developed countries reporting a prevalence of 10% of challenge-proven food allergies. Recognition of the Allergic March has played a crucial role in identifying causality in allergic conditions, linking atopic dermatitis to food allergy and food allergy to other atopic disorders, thereby highlighting opportunities in prevention and the importance of early intervention. This publication will establish the value of weaving the less well-understood, non-IgE-mediated food allergy into the Allergic March and mapping its progression through childhood and its associated co-morbidities. The proposed non-IgE-mediated Allergic March highlights the concomitant presentation of gastrointestinal symptoms and atopic dermatitis as early presenting symptoms in confirmed non-IgE-mediated allergies and the later development of atopic co-morbidities, including asthma and allergic rhinitis, similar to the IgE-mediated Allergic March. This publication highlights recent observations of a link between non-IgE-mediated food allergy in early childhood and functional gastrointestinal disorders in later life and also the reported occurrence of extra-intestinal manifestations at later ages. Although significant limitations exist in regard to the proposed evolution of the Allergic March model, the authors hope that this publication will influence the management of non-IgE-mediated gastrointestinal allergies and inform future research and interventions.
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Affiliation(s)
- Rosan Meyer
- Department Paediatrics, Imperial College, London, UK
| | - Adam T Fox
- Department of Paediatric Allergy, Evelina London Children's Hospital, London, UK
| | | | - Louise J Michaelis
- Department of Allergy and Immunology, Great North Children's Hospital, Newcastle Upon Tyne, UK
| | - Neil Shah
- Department Gastroenterology, Great Ormond Street Hospital, London, UK
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Dupont C. Food Protein-Induced Enterocolitis Syndrome and Proctocolitis. ANNALS OF NUTRITION AND METABOLISM 2019. [DOI: 10.1159/000493671] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 11/19/2022]
Abstract
Non-IgE-mediated, also labeled cell-mediated, allergic reactions to foods are more common than usually thought and probably account for approximately more than 40% of cases of cow’s milk allergy during infancy and young childhood. Food allergy is now described in the form of syndromes, among which food protein-induced enterocolitis syndrome (FPIES) and food protein-induced allergic proctocolitis (FPIAP) are gaining increased recognition. FPIES occurs in infancy but may also occur in older children and in adults. The dominant symptom is emesis, repetitive in the chronic FPIES form and explosive in the acute form. Acute FPIES begins 1– 4 h following ingestion of the offending food. Diarrhea is frequent, between 5 and 10 h later, and may be accompanied by lethargy and dehydration, which both characterize severity. Cow’s milk is the most frequent food trigger, followed by soy. FPIES may develop up to 1 year of age, but may also occur in the newborn, and is possible in exclusively breastfed infants, in relation with the mother’s consumption of offending foods. FPIES may occur to solid foods (grains like rice or oat, meats, fish, egg, and vegetables). When starting during infancy, FPIES has a good prognosis and disappears grossly at 2 years of age. FPIES to fish or shellfish is more frequent in older children and adults and is long lasting. International consensus guidelines for the diagnosis and management of FPIES have been published recently. FPIAP starts in the first few months of life and is typically manifested with rectal bleeding in well-appearing breastfed infants during the first months of life in reaction to cow’s milk consumed by the mother. The condition is transient but represents one of the major causes of colitis during infancy.
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Blackman AC, Anvari S, Davis CM, Anagnostou A. Emerging triggers of food protein-induced enterocolitis syndrome: Lessons from a pediatric cohort of 74 children in the United States. Ann Allergy Asthma Immunol 2019; 122:407-411. [PMID: 30742916 DOI: 10.1016/j.anai.2019.01.022] [Citation(s) in RCA: 54] [Impact Index Per Article: 9.0] [Reference Citation Analysis] [Abstract] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/11/2018] [Revised: 01/23/2019] [Accepted: 01/31/2019] [Indexed: 01/08/2023]
Abstract
BACKGROUND Food protein-induced enterocolitis syndrome (FPIES) is an infrequent non-IgE-mediated gastrointestinal allergic disorder that occurs mostly in infants and young children. FPIES food triggers vary among different geographic locations, and the condition is still underdiagnosed and underrecognized. OBJECTIVE To identify the triggers, characteristics, and management of FPIES in a pediatric US population of 74 children presenting to a tertiary center during a 3-year period. METHODS We performed a retrospective electronic record review of all pediatric patients with a diagnosis of FPIES who presented to Texas Children's Hospital emergency centers and clinics. RESULTS Most of our patients were white, and 65% had a positive family history of atopy. The median age at the first FPIES episode was 5 months (interquartile range, 4-6 months), and the median age at diagnosis was 11 months (interquartile range, 7-16 months). Grains (88%), cow's milk (49%), and vegetables (43%) were the most common food triggers in our cohort. Of the fruits, banana (24%) and avocado (16%) were predominantly reported. More than half of our patients experienced FPIES to multiple food triggers. CONCLUSION In our cohort, rice (53%) was the most common individual food trigger, surpassing cow's milk and soybean, previously reported as the most prevalent FPIES triggers in the United States. Banana (24%) and avocado (16%) rates were also much higher than in other studied populations, likely a reflection of different dietary and weaning habits in our area. Time from disease presentation to diagnosis was delayed, potentially because of difficulties in disease recognition. We noted a significant percentage of multiple-food FPIES in contrast to other populations.
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Affiliation(s)
- Andrea C Blackman
- Section of Pediatric Immunology, Allergy, and Rheumatology, Baylor College of Medicine, Houston, Texas; Department of Pediatrics, Section of Immunology, Allergy, and Rheumatology, Texas Children's Hospital, Houston, Texas
| | - Sara Anvari
- Section of Pediatric Immunology, Allergy, and Rheumatology, Baylor College of Medicine, Houston, Texas; Department of Pediatrics, Section of Immunology, Allergy, and Rheumatology, Texas Children's Hospital, Houston, Texas
| | - Carla M Davis
- Section of Pediatric Immunology, Allergy, and Rheumatology, Baylor College of Medicine, Houston, Texas; Department of Pediatrics, Section of Immunology, Allergy, and Rheumatology, Texas Children's Hospital, Houston, Texas
| | - Aikaterini Anagnostou
- Section of Pediatric Immunology, Allergy, and Rheumatology, Baylor College of Medicine, Houston, Texas; Department of Pediatrics, Section of Immunology, Allergy, and Rheumatology, Texas Children's Hospital, Houston, Texas.
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Food Protein-induced Enterocolitis Syndrome: Data From a Multicenter Retrospective Study in Spain. J Pediatr Gastroenterol Nutr 2019; 68:232-236. [PMID: 30320669 DOI: 10.1097/mpg.0000000000002169] [Citation(s) in RCA: 44] [Impact Index Per Article: 7.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/13/2022]
Abstract
OBJECTIVE The aim of the study was to describe clinical, epidemiological, and management characteristics of food protein-induced enterocolitis syndrome (FPIES) cases in Spain. PATIENTS AND METHODS Multicenter observational retrospective study. FPIES cases diagnosed in specialized units in Spain over 12 months in 2017 (January-December) according to the recently published international diagnostic criteria were included. RESULTS One hundred twenty patients (53.3% boys) were included. The majority were acute cases (111) with mild-to-moderate severity (76.7%). Triggering foods were cow's milk (48/120), fish (38), egg (13), rice (12), and soy (1). The majority (84.2%) of the patients had FPIES to 1 food only. In addition to vomiting (100%), pallor (89.2%), and altered behavior (88.3%) were most frequently observed in acute forms. On the contrary, diarrhea (70%), abdominal distension (33.3%), and blood in stools (44.4%) were more frequently observed in chronic cases. Oral challenge was performed in 18.9% of the acute forms compared to 44.4% of the chronic forms. The most common treatment was intravenous fluids followed by ondansetron. Corticosteroids were used in 6 patients (5 with acute symptoms and 1 chronic). Seven patients were treated with antibiotics for suspicion of infection. Most cases of cow's milk FPIES were treated with extensively hydrolyzed formulas (69.8%). CONCLUSIONS FPIES is not uncommon in our units. Unlike other published series, fish and egg are important triggers in our country. A greater knowledge and diffusion of the international consensus criteria will allow a better characterization of the cases and a standardization of their management.
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Wang KY, Lee J, Cianferoni A, Ruffner MA, Dean A, Molleston JM, Pawlowski NA, Heimall J, Saltzman RW, Ram GS, Fiedler J, Gober LM, Spergel JM, Brown-Whitehorn TF. Food Protein–Induced Enterocolitis Syndrome Food Challenges: Experience from a Large Referral Center. THE JOURNAL OF ALLERGY AND CLINICAL IMMUNOLOGY-IN PRACTICE 2019; 7:444-450. [DOI: 10.1016/j.jaip.2018.09.009] [Citation(s) in RCA: 24] [Impact Index Per Article: 4.0] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Received: 04/27/2018] [Revised: 09/06/2018] [Accepted: 09/09/2018] [Indexed: 10/28/2022]
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Jacob S, Vitor AB. Cow's milk protein intolerance imitating septic shock in a young infant. ANALES DE PEDIATRÍA (ENGLISH EDITION) 2019. [DOI: 10.1016/j.anpede.2018.03.001] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/30/2022] Open
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Jacob S, Bonito Vitor A. Intolerancia a la proteína de la leche de vaca simulando shock séptico en un lactante pequeño. An Pediatr (Barc) 2019; 90:52-53. [DOI: 10.1016/j.anpedi.2018.01.020] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/07/2017] [Revised: 12/15/2017] [Accepted: 01/11/2018] [Indexed: 10/17/2022] Open
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Food Protein-Induced Enterocolitis Syndrome (FPIES): Review of Recent Guidelines. Curr Allergy Asthma Rep 2018; 18:28. [PMID: 29623454 DOI: 10.1007/s11882-018-0767-9] [Citation(s) in RCA: 9] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/17/2022]
Abstract
PURPOSE OF REVIEW To increase understanding of food protein-induced enterocolitis syndrome (FPIES), a non-immunoglobulin E (IgE)-mediated reaction to food, by reviewing a growing body of literature, including recently published international consensus guidelines. RECENT FINDINGS FPIES primarily affects infants and young children and is characterized by the delayed onset of gastrointestinal symptoms, predominantly repetitive vomiting, in response to a trigger food. Symptoms are often severe and can lead to shock. Diagnosis can be challenging due to a wide differential diagnoses and lack of disease biomarkers. FPIES is a clinical diagnosis, with allergy testing playing a very limited role, if any. Medically supervised oral food challenges are used to monitor resolution of disease, which generally occurs in early childhood. FPIES is an important condition presenting to clinicians in a variety of settings. Recent international consensus guidelines and a growing body of literature can better equip practitioners to care for these often-challenging patients.
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Ribeiro A, Moreira D, Costa C, Pinto Pais I. Food protein-induced enterocolitis syndrome: a challenging diagnosis. BMJ Case Rep 2018; 2018:bcr-2017-222822. [PMID: 29437805 DOI: 10.1136/bcr-2017-222822] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/03/2022] Open
Abstract
Food protein-induced enterocolitis syndrome (FPIES) is a non-IgE-mediated gastrointestinal food hypersensitivity triggered by food proteins. It may present acutely, with repetitive vomiting, diarrhoea and lethargy leading to dehydration and eventually shock or insidiously with intermittent emesis, chronic diarrhoea or failure to thrive. We describe a paediatric male patient with recurrent sepsis-like episodes of fever, lethargy, ashen-grey skin colouration and vomiting followed by diarrhoea. These episodes were triggered by cow's milk formula and grains. Laboratory tests revealed leucocytosis, thrombocytosis, metabolic acidosis and elevated C reactive protein. After exclusion of other differential diagnoses, the diagnosis of FPIES was established on clinical improvement with withdrawal of the offending food and positive oral food challenge. FPIES diagnosis requires a high index of suspicion and is frequently delayed, which contributes to an increased morbidity. This is due to the wide spectrum of clinical presentations and due to the absence of specific diagnostic tests.
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Affiliation(s)
- Andreia Ribeiro
- Department of Paediatrics, Hospital Center of Vila Nova de Gaia/Espinho, Vila Nova de Gaia, Portugal
| | - Diana Moreira
- Department of Paediatrics, Hospital Center of Vila Nova de Gaia/Espinho, Vila Nova de Gaia, Portugal
| | - Cristina Costa
- Pediatric Gastroenterology, Centro Hospitalar Gaia Espinho, Vila Nova de Gaia, Portugal
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